Author Archives: faoj

2021 ANNOUNCEMENT

Dear authors and readers,

The Foot and Ankle Online Journal (FAOJ) has just wrapped up it’s 13th year of providing open access, peer-reviewed articles to the international foot and ankle medical community. The journal grew from the roots of a blog by Dr. Al Kline, titled the Podiatry Internet Journal. The blog quickly expanded to become the Foot and Ankle Online Journal within its first year of publishing, with a mission of providing open-access peer-reviewed case reports. As the journal continued to grow and gain readership, it was adopted as the official journal of the International Foot & Ankle Foundation for Education and Research in 2008.

As of this writing, the journal has published over 500 articles, sees approximately 40,000 readers per month, and accepts a variety of submissions including case studies, meta-analyses, and original research articles. Just as the Podiatry Internet Journal had outgrown its utility, the editorial board of the FAOJ has made the decision to retire the journal to put its resources towards a new journal, titled the Journal of the International Foot & Ankle Foundation. This new journal is anticipated to launch by the end of 2021. The FAOJ will continue at this webspace as an archive.

The final issue of the FAOJ was published at the end of 2020, and the journal will therefore no longer be accepting new submissions. If you would like to make a submission to the upcoming Journal of the International Foot & Ankle Foundation, please e-mail e-mail the editor for submission guidelines, although you should be aware that there is not yet a confirmed publication date for the first issue.

We sincerely thank you for your past support and contributions and look forward to your participation with IFAF’s new journal in the future.

Craig E. Clifford, DPM, MHA, FACFAS, FACPM
Editor

G. Dock Dockery, DPM, FACFAS
Editor-in-Chief

Management of an open crush fracture to the foot from a lawnmower injury: A case report

by Rosario Saccomanno, DPM1*, Matthew S. Kalmar, DPM1

The Foot and Ankle Online Journal 13 (4): 14

A 50-year-old male who reported to the emergency department (ED) with an open fracture to his right great toe sustained from a lawnmower injury is presented. The patient underwent emergent overnight podiatric surgery for treatment of the traumatic crush injury. Removal of all non-viable skin, soft tissue, and bone was performed intra-operatively which ultimately resulted in a partial right hallux amputation with primary closure. This case report aims to highlight the accepted treatment protocols set forth in the management of pedal open crush injuries. Open fractures are true podiatric emergencies and delay in treatment places the patient at increased risk for poorer prognosis.

Keywords: open fracture, trauma, crush, partial amputation

ISSN 1941-6806
doi: 10.3827/faoj.2020.1303.0014

1 – Huntington Hospital at Northwell Health, Huntington, N.Y.
* – Corresponding author: saccoross@gmail.com


A pedal fracture is considered “open” when broken bone is immediately exposed to the atmosphere [1]. The mechanism of injury causing such injury is typically that of high velocity, crush-type trauma powerful enough to penetrate the layers of the integumentary system to cause said fracture. These injuries are true podiatric emergencies that are often accompanied by severe bone comminution and/or complex soft tissue degloving and may be constituted in polytrauma cases where primary, secondary, and tertiary surveys of the patient are imperative [2]. The essential goals in the management of open fractures revolve around prevention of infection, establishment of bony union, and restoration of function [1,3]. Treatment calls for non-excisional and/or excisional debridement, tetanus and antibiotic prophylaxis, and fracture stabilization, all of which have a synergistic role in attempting to prevent sepsis, minimize disability, and promote limb salvage [4].

Wound lavage acts as a form of non-excisional mechanical debridement to eradicate foreign debris from the wound site and to dilute the concentration of bacteria to a burden less than that of 105 in attempt to prevent infection [5]. Irrigation may be performed with low- or high-pressure modalities. Irrigation may be in the form of 0.9% sterile saline solution or agents such as povidone-iodine, chlorhexidine, and/or hydrogen peroxide which introduce antisepsis to the wound site [6,7]. Excisional debridement goes hand-in-hand with irrigation to further mechanically remove foreign material and nonviable tissue from the wound site [1]. Removal of devitalized tissue prepares the wound site for healthy tissue demarcation which will allow for wound closure to occur in the absence of bacterial bioburden [4].

Open fractures are particularly vulnerable to deep wound polymicrobial infection. Empiric antibiotic selection should be based on wound size, extent of soft tissue injury, and type of environment in which the injury was sustained [1,4]. Gustilo and Anderson devised a classification system with 3 types of open fractures which serves as a guide for recommended antibiotic coverage [8]. Type I open fractures are less than 1 cm in wound diameter with a simple fracture pattern and adequate soft tissue coverage. Type II open fractures have a wound diameter between 1 and 5 cm(s) with moderate soft tissue disruption yet intact periosteal and soft tissue coverage. Type III open fractures have extensive wound diameters greater than 5 cm(s) with comminution, extensive soft tissue damage, or traumatic amputation and can be further subdivided into those injuries with adequate soft tissue coverage (Type III-A), inadequate soft tissue coverage (Type III-B), or arterial compromise (Type III-C) [4,8].

Appropriate tetanus prophylaxis is imperative in cases of open fractures due to their inherently high risk of infection by Clostridium tetani, an obligate spore-forming anaerobe commonly found in farm environments that produces tetanospasmin. This is an exotoxin that causes spastic paralysis of voluntary muscles and can lead to fatal cases of respiratory arrest [1]. The Centers for Disease Control and Prevention (CDC) set forth recommendations in tetanus prophylaxis for wound management [9]. For those individuals sustaining an injury in a contaminated environment, both Tetanus-Diphtheria-Pertussis (TdaP) or Tetanus toxoid (Td) and Tetanus Immunoglobulin (TIG) are warranted for patients with an unknown vaccination history, those with fewer than 3 doses of Tetanus Toxoid (TT), or in cases where it has been more than 5 years since the patient’s last dose of TT. For clean, minor wounds, TdaP or Td are indicated if the patient has an unknown vaccination history or has received less than 3 doses of TT. TdaP or Td is recommended for those patients who have had 3 or more doses of TT if more than 10 years has elapsed since receiving the last dose of TT [9].

Regarding fracture stabilization, reduction followed by immobilization may be necessary to perform in the acute ED setting for open crush injuries. In the operative setting, external fixators are often employed to hold fractured fragments out to anatomic longitudinal length while not interfering with open wound management in cases of contaminated or infected open fractures. Fixation using either open or percutaneous Kirschner (K) Wires are often employed to achieve internal splintage of fracture fragments [4]. It is recommended that the implementation of internal hardware such as that of plates and/or screws is reserved for use once the open fracture site is deemed clean and the site can be primarily closed, which may need to occur in a staged-procedure fashion [1].

Case Report

In the late evening of August 23, 2018, a 50-year-old male with a past medical history significant for alcoholism presented to the ED after his right foot was run over by a commercial lawnmower during farm and landscaping work earlier that afternoon. The patient reported 6 hours from the time of injury to presentation to the hospital. The patient reported wearing steel-toe boots during the incident and reported that he continued to work through his shift after sustaining the injury. The patient denied any trauma to the left foot or any constitutional symptoms upon presentation to the ED.

After thorough primary and secondary examinations were performed, a focused lower extremity physical examination noted a diffusely edematous and ecchymotic right foot with palpable peripheral pulses and soft and non-tender compartments. Delayed capillary refill time to the right first digit with absent protective and light touch sensation were noted. A traumatically amputated distal tuft of the right hallux revealed a Gustilo-Anderson Type III-A open fracture immediately exposing segmental comminution to the distal aspect of the distal phalanx along with gravel debris and active bleeding to the site. The right hallucal nail plate was found to be loose with subungual hematoma encompassing 100% of the underlying nail bed. The patient was unable to passively move the right first toe when prompted.

The nature, severity, and time sensitivity of the patient’s injury called for immediate bedside management of his open crush fracture in the ED setting. Cefazolin, ciprofloxacin, and penicillin G potassium were administered and the patient was started on intravenous fluids. The patient was given an intramuscular (IM) dose of 0.5 mL TdaP for tetanus prophylaxis as he admitted it had been more than 10 years since his last tetanus booster. The open fracture was copiously irrigated using a total of 9 liters of 0.9% sterile saline solution. A right hallux anesthetic block was then administered. An excisional debridement of nonviable tissue as well as a total hallucal nail avulsion to evacuate subungual hematoma and to assess the true extent of the injury were performed (Figure 1). X-ray confirmed comminution of the distal phalanx with surrounding soft tissue swelling (Figure 2). The right hallux was immobilized by means of a basket-weave splint for fracture stabilization and the right foot was dressed with a bulky, compressive dressing. The patient was given pain medication for pain control.

Figure 1 Preoperative image demonstrating open fracture of the right foot with traumatic amputation of the hallucal distal tuft.

Figure 2 Plain dorsoplantar, oblique, and lateral radiographs of the foot revealing a comminuted fracture of the hallucal distal phalanx with surrounding soft tissue swelling.

At this time, the patient was prepped for emergency surgery and was instructed for his subsequent admission. The patient was booked for removal of nonviable skin, soft tissue, and bone from the right foot and instructed on the possible need for a staged procedure. The patient consented to the proposed operation and medical clearance was granted. The patient underwent the emergent operation overnight under monitored anesthesia care with local anesthetic.

Figure 3 Postoperative plain dorsoplantar, oblique, and lateral radiographs of the foot illustrating salvage of the proximal 1/3 base of the distal phalanx resulting in a partial hallux amputation.

Degloved, nonviable skin and soft tissue of the right hallux was excisionally debrided and all comminuted fragmentation of the exposed distal phalanx was excised and passed off for pathological analysis. The proximal 1/3 base of the hallucal distal phalanx was found to be viable and was salvaged intra-operatively (Figure 3). The surgical site was irrigated with 9 liters of 0.9% sterile saline solution. The remaining base of the distal phalanx was rasped to remove sharp edges and bone wax was applied over its surface. Primary closure was achieved using a flap from the plantar hallucal skin and soft tissue which ultimately resulted in a partial hallux amputation.

Surgical pathology revealed findings of hemorrhage and reactive changes without any evidence of bone or soft tissue infection. The patient was continued on IV antibiotics until the date of hospital discharge when he was transitioned to a 10-day course of cephalexin per the infectious disease specialist. The patient healed uneventfully on the outpatient basis with appropriate return to normal shoewear.

Discussion

In this case report, a farm injury caused by a commercial lawn mower resulted in an open fracture of the right hallux that required emergent overnight surgery. Immediate bedside irrigation was initiated using an initial 9 liters of 0.9% sterile saline solution to remove gross debris and bacterial contamination followed by another 9 liters of the same solution used in the operating room. Anglen identified volume, pressure, and pulsation as three variables as part of a proposed irrigation protocol utilized for the treatment of open fractures, with his recommendation calling for 3 liters of irrigation used for Type I open fractures, 6 liters for Type II open fractures, and 9 liters for Type III open fractures [6]. Though antiseptic agents have been purported to have bactericidal properties, they have been reported to cause cytotoxicity, impaired osteoblast function, delayed wound healing, and chondrocyte damage; hence, 0.9% sterile saline solution is considered the standard of choice irrigant in cases of open fractures [1]. Regarding the pressure variable in the irrigation protocol, Anglen and colleagues in another study observed a 100-fold decrease in slime-producing Staphylococcus bacterial count in wounds treated with pulsatile lavage as compared with bulb irrigation when the same solutions and volumes were used in both delivery systems [7]. Anglen reported that even though pulsation may in theory remove surface debris by means of tissue elasticity, there is no established recommendation due to limited studies on this irrigation variable [6].

Another important consideration in the management of open fractures is timing from onset of injury to debridement. The American College of Surgeons Committee on Trauma (ACS-COT) deems debridement within 6 hours from time of injury to be the standard of care as a time lapse greater than this can result in 1 g of tissue inoculated with a single bacterium to duplicate 105 bacteria to convert a contaminated wound to an infected wound [4,5,10]. More recent clinical data in the literature lacks support of this postulation and concludes that early debridement is not an independent predictor of decreased risk of infection [11]. In this case, non-excisional and excisional debridement were performed at bedside within 6 hours from the onset of the patient’s injury and shortly thereafter in the operative setting, which allowed for primary wound closure via a plantar hallucal skin and soft tissue flap.

Tetanus and antibiotic prophylaxis were initiated in this case due to the severity of the patient’s open fracture as well as the farm environment where it occurred. The patient was given an IM dose of TdaP per the ED physician as more than 10 years had elapsed since the patient’s last tetanus booster. The rate of bone and/or soft tissue infection following an open fracture is purported to be 3-25% with the most common pathogens being that of natural skin flora, namely coagulase-positive Staphylococcus aureus [1,8]. For the patient’s Gustilo-Anderson Type III-A open fracture, the ED physician administered cefazolin, ciprofloxacin, and penicillin G potassium per recommendation of the podiatry service which provided appropriate gram-positive, gram-negative, and anaerobic coverage. With Type III-A open fractures occurring in a farm environment, penicillin G potassium is warranted for Clostridium spore coverage [1].In a study of 1104 open fractures, Patzakis and Wilkins reported that early administration of antibiotics, deemed within 3 hours of onset of injury, was the single most important factor in reducing rate of infection (4.7% compared with 7.4% for those that received antibiotics after 3 hours from time of injury) [12]. Following surgical debridement and primary closure, it is recommended that antibiotics are continued for an additional 24 to 48 hours in the post-operative period [1]. In this case, bone and soft tissue of the right hallux that was passed off for pathological analysis came back negative for evidence of infection. The infectious disease specialist continued the patient on cefazolin in the immediate post-operative period and discharged him with a 10-day course of cephalexin due to the open and contaminated nature of the wound upon initial patient presentation.

The emergent nature of the patient’s open fracture ultimately resulted in a partial hallux amputation. Due to the extensive amount of comminution to the distal aspect of the distal phalanx bone, these fragments could not be apposed in an improved position with internal or external fixation and were therefore deemed non-salvageable and excised altogether. Rasping of the salvaged 1/3 base of the hallucal distal phalanx and implementation of bone wax were used to smoothen osseous contour and deter spicule regrowth, respectively. Primary closure via use of a plantar hallucal skin and soft tissue flap was achieved as the site was deemed clean as a result of timely irrigation and debridement from onset of injury.

Conclusion

This article highlights a case of an open fracture to the great toe which resulted in a partial hallux amputation and outlines the accepted management of such traumatic injuries through a review of literature. Open fractures are true podiatric emergencies that can pose life-threatening changes to patients and their well-being. Traumatic great toe amputations may lead to functional and psychological disability as well as decreased propulsive effort during the push-off phase of the gait cycle. Prognosis is highly dependent on both timing of patient presentation to seek medical attention as well as on prompt rendering of medical care by the practitioner. If treatment is delayed, the patient risks suffering from a greater likelihood of morbidity and poorer prognosis. By increasing the knowledge and awareness of these debilitating injuries and their treatment protocols, the patient will be best served and the practitioner will be best guided in the overall aim to prevent infection, minimize disability, and promote limb salvage.

References

  1. Sexton SE: Open fractures of the foot and ankle. Clin Podiatr Med Surg. 2014 Oct;31(4):461-86.
  2. Biffl W, Harrington D, Cioffi W: Implementation of a tertiary trauma survey decreases missed injuries. J of Trauma: Injury, Infection, and Critical Care 2003 Jan;54(1):38-43.
  3. Weitz-Marshall AD, Bosse MJ: Timing of closure of open fractures. J Amer Acad Ortho Surgeons Nov-Dec 2002;10(6):379-84.
  4. McGlamry ED: “Open Fractures,” in McGlamry’s Comprehensive Textbook of Foot and Ankle Surgery, Vol 2, edited by JT Southerland, p 1499, Philadelphia, Lippincott Williams & Wilkins, 2013.
  5. Bowler PG: The 10(5) bacterial growth guideline: reassessing its clinical relevance in wound healing. Ostomy Wound Manage. 2003 Jan;49(1):44-53.
  6. Anglen JO: Wound irrigation in musculoskeletal injury. J Amer Acad Ortho Surgeons Jul-Aug 2001;9(4):219-26.
  7. Anglen JO, Apostoles S, Christensen G, et al: The efficacy of various irrigation solutions in removing slime-producing Staphylococcus. J Ortho Trauma 1994 Oct;8(5):390-6.
  8. Gustilo RB, Anderson JT: Prevention of infection in the treatment of one thousand and twenty-five open fractures of long bones. J Bone and Joint Surg. 1976 Jun;58(4):453-8.
  9. Centers for Disease Control and Prevention: Tetanus – for clinicians. Available at: (https://www.cdc.gov/tetanus/clinicians.html). Accessed February 16, 2020.
  10. American College of Surgeons: Resources for optimal care of the injured patient 2014/resources repository. Available at: (https://www.facs.org/quality-programs/trauma/tqp/center-programs/vrc/resources). Accessed February 15, 2020.
  11. Pollak AN: Timing of débridement of open fractures. J Amer Acad Ortho Surgeons 2006;14(10 Spec No.):S48-51.
  12. Patzakis MJ, Wilkins J: Factors influencing infection rate in open fracture wounds. Clin Ortho and Rel Research 1989 Jun;(243):36-40.

 

The clinical and patient centered outcomes following surgical correction of tailor’s bunion in an acute hospital based podiatric surgery service

by Syarifah Wilson1*, BSc. MSc. MChS.; James Cowden1 BSc. MSc. MChS. FCPodS

The Foot and Ankle Online Journal 13 (4): 13

This paper presents the results of the clinical and patient reported outcomes of patients following fifth metatarsal scarf osteotomy performed for correction of tailor’s bunion deformities utilizing the Manchester Oxford Foot Questionnaire (MOXFQ), Patient Satisfaction Questionnaire-10 (PSQ-10) and radiographic analysis. The electronic records of 24 patients (25 feet) were reviewed retrospectively after they had undergone the procedure between 2014 and 2018. Student paired t-test was used to compare pre- and postoperative outcomes from the MOXFQ and fourth-fifth intermetatarsal angle (4-5 IMA) and fifth metatarsophalangeal joint angle (5-MTPA). Differences were considered statistically significant if the probability of the null hypothesis was less than 0.05. A 95% confidence interval was observed for MOXFQ and radiographic measurements. For PSQ-10, a thematic analysis was undertaken to identify patterns of important themes in question 1 responses data and a quantitative descriptive design was applied to question 2 to 10 numerical data scores. A significant reduction in all three MOXFQ domains was observed: scores of walking/standing (W/S) 54.7 to 20.2; pain (P) 60.4 to 22.2 and social interaction (SI) 50.4 to 6.7. A 95% confidence interval for the difference was given for MOXFQ scores: 22.786, 46.254 (W/S); 29.507, 46.893 (P) and, 34.534, 52.826 (SI). The mean preoperative 4-5 IMA was 11.6° and 5-MTPA measurement was 19.8°; the mean postoperative 4-5 IMA was 5.7° and 5-MTPA was 8.0°. The p-values for the W/S, P and SI differences and radiographic evaluations were <0.001. PSQ-10 scored 88.0 and 96% recorded ‘pain relief’ in patient’s expectation. There were 6 cases (24%) of surgical sequelae. This service review indicated high levels of patient satisfaction with the procedure and a relatively low number of complications. From a clinical point of view, it allows a greater amount of correction in transverse and sagittal planes variants and is inherently stable amenable to two screws fixation.

Keywords: tailor’s bunion, scarf osteotomy, MOXFQ, PASCOM-10, PSQ- 10

ISSN 1941-6806
doi: 10.3827/faoj.2020.1304.0013

1 – Sheffield Teaching Hospitals NHS Foundation Trust
* – Corresponding author: Syarifah.Wilson@nhs.net


Tailor’s bunion, also known as bunionette or digitus quintus varus, is a pathological deformity of the fifth metatarsophalangeal joint (MTPJ), characterised by a bony protuberance of the lateral aspect of the fifth metatarsal head [10]. The classification of tailor’s bunion by Coughlin in 1991 is based on standard weight-bearing radiographic measurements [8]. Coughlin [14] defined the three types of tailor’s bunion as:

  • Type 1: A lateral exostosis of the fifth metatarsal head (dumbbell-shaped)
  • Type 2: Lateral bowing of the distal aspect of the fifth metatarsal shaft
  • Type 3: Increased fourth-fifth intermetatarsal angle (4-5 IMA), of more than 10° for patients with symptomatic tailor’s bunion
  • Type 4: Added on by DiDomenico in 2013, describes patients with a combination of deformities; including 2 or more combinations of the above.

The pathophysiology of tailor’s bunion is multifactorial attributed to both its anatomical and biomechanical variations and therefore, understanding and characterising each component of the deformity is key to treating it successfully [24]. Although tailor’s bunion is not analogous to hallux abducto valgus (HAV) in its etiopathogenesis, both deformities can frequently occur concomitantly; known as splayfoot deformity [17,36]. However, the diagnosis of tailor’s bunion is often overlooked when patients present with HAV as their main complaint [13].

First line conservative treatments of the pathology include footwear alteration, use of orthoses, oral/topical analgesics and corticosteroid injection [43]. Surgical intervention may be considered if symptoms remain persistent despite conservative management and/or when the disease progresses to its more severe form. Different procedures have been suggested and reported for the tailor’s bunion with varying outcomes. Similar to the surgical management of HAV, osteotomies of the fifth metatarsal can be grouped into proximal, mid shaft or distal, depending on the severity of deformity [3]. Some of these corrective measures range from a simple lateral fifth metatarsal head exostectomy for early stages of the deformity to osteotomies and lastly, in cases of unreconstructable deformities may require a fifth metatarsal head resection [34,40].

As with any surgical procedure, there are advantages and disadvantages. For less severe tailor’s bunion deformities, distal osteotomies are beneficial and, owing to the increased blood supply at this location, have lesser risk of delayed and/or non-union [29]. In contrast, proximal osteotomies are effective in correcting deformities with a 4-5 IMA which exceeds 9° and provides greatest satisfaction score but disadvantages would include inherent instability of the location of the osteotomy, disruption of intra- and extraosseous blood supply of the metatarsal, and technical demand [43]. Similar to proximal osteotomies, diaphyseal osteotomies have also been found to achieve a greater 4-5 IMA correction and allows a triplanar correction [5]. Furthermore, diaphyseal osteotomies have more bone to bone surface thereby allowing fixation without compromising the vascular supply [5,25].

Procedure

Within the Sheffield Teaching Hospital National Health Services Foundation Trust (STH NHSFT) Department of Podiatric Surgery, the fifth metatarsal scarf osteotomy is routinely performed for the correction of tailor’s bunions of varying intermetatarsal angles. It is a diaphyseal osteotomy originally developed by Weil as the “Reverse scarf” and later popularised by Barouk [2,42]. The procedure in STH NHSFT was performed by three podiatric surgeons; a Consultant and two Registrars. The two Registrars had undertaken their surgical training with the Consultant and it is therefore hoped that this would minimise the commonly occurring variations in surgical approach/technique [32].

The surgical technique performed is similar to that described by Maher and Kilmartin (2010) whereby the osteotomy is fixed with two countersunk 2.0mm cortical screws (Figure 1) [25].

Figure 1 Pre- and postoperative demonstration of surgical technique.

Summary of Surgical Technique

  1. The procedure is carried out under regional anesthesia block with the use of an ankle tourniquet.
  2. A linear longitudinal skin incision is made down the lateral border of the fifth metatarsal, with a double elliptical incision over the fifth metatarsal head.
  3. Sharp and blunt dissection is undertaken maintaining hemostasis.
  4. Double elliptical lateral capsular incision is performed.
  5. The tissue is reflected from the dorsal and plantar metatarsal and the fifth MTPJ exposed.
  6. Lateral eminence is removed with a sagittal saw.
  7. A longitudinal cut to the metatarsal shaft is made with a slight dorsal inclination through both the lateral and medial cortices.
  8. A perpendicular cut is made transversely across the metatarsal dorsally at the distal end.
  9. An oblique cut is made transversely across the metatarsal plantarly at the proximal end.
  10. The metatarsal head is internally rotated about a proximal axis whilst preserving its length and temporarily held with a bone clamp.
  11. When satisfied with the position, two bicortical holes are drilled, countersunk and measured.
  12. Permanent fixation is applied utilizing two DePuy Synthes 2.0mm cortical screws.
  13. Stability is assessed versus distraction in all planes.
  14. Lateral overhang is removed with a sagittal saw.
  15. Deep tissue closure is undertaken with 3/0 vicryl and the skin with a 4/0 monocryl.
  16. Dressings are applied and postoperative shoe worn. Patients are allowed to partially ambulate on the heel of the operated foot with crutches for 2 weeks.
  17. Patients are reviewed at 2 weeks postoperatively for suture removal and transfer into supportive footwear.

Materials and Methods

This was a single-center retrospective service evaluation which reviewed the outcome of 24 patients (25 feet) who underwent a fifth metatarsal scarf metatarsal osteotomy between 2014 and 2018. This paper utilizes existing surgical audit data generated by and extracted from the Podiatric Audit of Surgery and Clinical Outcome Measurement (PASCOM-10). This online database is registered to the STH Podiatric Surgery Unit. Any PASCOM-10 reports with incomplete Manchester Oxford Foot Questionnaire (MOXFQ) and Patient Satisfaction Questionnaire (PSQ-10) data have been excluded.

The two functional outcome scoring instruments used within PASCOM-10 for this service evaluation were:

i) the MOXFQ; captured on the day of surgery and, at six months’ final check postoperative appointment.

ii) the PSQ-10, taken at the final six months follow up (no pre-treatment component for this domain).

Both MOXFQ and PSQ-10 are two powerful Patient Reported Outcome Measures (PROMs) instruments as they provide an insight into patient experiences of pain and foot function as well as an overall indication of the surgical outcomes [28]. The MOXFQ contains 16 items, each with five response options consisting of three underlying domains: Walking/Standing (W/S) (seven items), Pain (P) (five items), and Social Interaction (SI) (four items). Similar to a Likert scale, each item response is scored from 0 to 4; where the highest value denotes the most severe state and therefore, lower scores in each domain in the postoperative MOXFQ are indicative of positive patient outcomes [11]. The three domains scales have gone through extensive testing and have shown to have excellent psychometric properties in terms of reliability, validity and responsiveness, and is comparable to other known instruments such as American Orthopaedic Foot and Ankle Score (AOFAS) and Short-Form health survey questionnaire-36 [11,12].

In comparison to the MOXFQ, the PSQ-10 has yet to be formally tested for its validity, however, the instrument has been chosen for use in PASCOM-10 due to its reliability and repeatability [27,39]. The PSQ-10 asks patients a series of 10 questions relating to their experience of an episode of care. In question 1, it outlines the reason why the patient had sought treatment from the services. For this section of the questionnaire, there is no scoring attached and therefore, not included in the final scoring of the patient satisfaction. For question 2 to 10, these questions are in a fixed-response format with a maximum score of 100 to indicate satisfaction while scores below 70 are indicative of poor surgical outcomes and patient satisfaction [45].

A quantitative descriptive design was applied to the numerical data scores obtained from MOXFQ and PSQ-10 question 2 to 10 and radiographic evaluations. This allows for application of student paired t-test utilizing IBM Statistical Package for Social Sciences (SPSS) 24.0 to compare the MOXFQ as well as fifth MTPJ angle (5-MTPA) and 4-5 IMA measurements pre- and 6 months postoperative. The first question of the PSQ-10 is distinguished from the rest of the questionnaire as it incorporates a free text response space for patients to articulate their expectations from the treatment. Therefore, a qualitative approach was required for further analysis of the answers and to identify patterns of important themes in the responses data. Incorporation of this qualitative analysis of such statements contributes to the evaluation of the service [1].

Essential data collection on PASCOM-10 begins when patients are listed for surgical treatment. The start of an episode commences with a referral and finishes with a discharge [6]. Each patient may require multiple episodes recorded and, in the event of a revision surgery, a new treatment episode would be required. Should there be any postoperative events such as sequela i.e., complications, clinicians are required to record these data onto PASCOM-10 as part of their routine practice. The data metrics used for this study have all been obtained from patients exclusive to the STH Podiatric Surgical unit; who have all been informed of, and consented to their data being potentially used for educational or research purposes. Consenting patients would complete their MOXFQ and PSQ-10 forms before the data is transferred onto PASCOM-10. To ensure unsolicited answers are obtained, the questionnaires are generally completed in the outpatient waiting room following the consultation whereby they were listed for surgery.

For the MOXFQ scores, a hypothesis test was set up as the following below;

● H0 (null): There is no effect of the procedure on MOXFQ W/S,P and SI

● H1 (alternate): There is an effect of the procedure on MOXFQ W/S, P and SI

The decision to reject the null hypothesis (H0) or fail to reject was based on the p-value. Differences were considered statistically significant if the probability of the null hypothesis given the data was less than 0.05.

Figure 2 Pre- and postoperative A/P radiograph of fifth metatarsal scarf osteotomy with concurrent procedure scarf and Akin osteotomy.

A 95% confidence interval was observed for MOXFQ as it provides a statement on the level of confidence that the true value for a population lies within a specified range of values [38].

Radiographic Data Collection

Anteroposterior (A/P) weight-bearing radiographic measurements were obtained at pre- and postoperatively at 2 weeks. The radiographic evaluation, reviewed by the first author, compared the pre- and postoperative 5-MTPA and the 4-5 IMA. The 5-MTPA was measured by bisecting the fifth metatarsal shaft and the shaft of the fifth proximal phalanx and the 4-5 IMA was measured utilizing the traditional technique of bisecting the long axes of the fourth and fifth metatarsal shafts. These measurement techniques were preferred as they were found to be both reliable and reproducible in comparison to others such as the modified version by Fallat & Buckholz [16,37]. The average 4-5 IMA in normal patients has been reported to be 6.4 to 9.1 degrees and 8.7 to 10.8 degrees in symptomatic tailor’s bunion [9,30]. The 5-MTPA was determined to be 10.2° varus in normal feet and 16.6° varus in symptomatic tailor’s bunions [30].

It is recognised and accepted by the study that the preoperative x-ray films are weightbearing and the postoperative are non-weight bearing. This could lead to an inaccuracy in the angular improvement in the surgery which is one of the shortfall of this aspect of the study.

Results

At the time of surgery, as displayed on Table 1, the mean age was 44; range 19-74 years old (y/o). 21 (84%) patients were female, and 4 (16%) were male. The majority of the sample were females (N=21) with an average age of 43.6 y/o (1.dp) and a standard deviation (SD) of 17.9. There were 4 males who had the procedure with an average age of 44.5 y/o (1.dp) and SD of 24.3. An age range of 25-29 y/o showed to have the highest percentage (20%) of patients with 4 females and 1 male.

Age Range Male (N) Male (%) Female (N) Female (%) Total (N) Total (%)
15-19 0 0% 2 8% 2 8%
20-24 1 4% 1 4% 2 8%
25-29 1 4% 4 16% 5 20%
30-34 0 0% 1 4% 1 4%
35-39 0 0% 0 0% 0 0%
40-44 0 0% 3 12% 3 12%
45-49 0 0% 3 12% 3 12%
50-54 1 4% 2 8% 3 12%
55-59 0 0% 0 0% 0 0%
60-64 0 0% 2 8% 2 8%
65-69 0 0% 0 0% 0 0%
70-74 1 4% 3 12% 4 16%
Total 4 8% 21 84% 25 100%
Average 44.50

≈ 45

43.62

≈ 44

43.76

≈ 44

Table 1 Patient demographics.

Table 2 demonstrates that 5 patients underwent additional procedures at the same time as the tailor’s bunion repair. One patient (4%) had one concurrent procedure and 4 patients (16%) had two additional concurrent procedures. These additional procedures included HAV repair by scarf and Akin osteotomies; repair of lesser toes deformities: hammer toes by 2nd, 4th and 5th digit excisional arthroplasty and a Lapidus procedure for treatment of concurrent hypermobile HAV. Patients’ health status was summarised by the American Society of Anaesthesiologists (ASA). ASA grade 1 accounted for 80% of patients and ASA grade 2 accounted for the remaining 20% which suggests that the patients who had the procedure were generally normal and healthy with only mild systemic diseases.

Count (N) Count (%)
Total no. of procedures 25 100%
ASA Grade 1 20 80%
ASA Grade 2 5 20%
Patients receiving ≥ procedure 5 20%
Patient (s) receiving 1 concurrent procedure 1 4%
Patient (s) receiving 2 concurrent procedure 4 16%
Additional concurrent procedure (s):
scarf and Akin osteotomy 3 12%
Arthrodesis 1st MTPJ 1 4%
Excisional arthroplasty 2nd digit 2 8%
Excisional arthroplasty 4th digit 1 4%
Excisional arthroplasty 5th digit 1 4%
Lapidus 1st metatarsal-cuneiform joint 1 4%

Table 2 Summary of procedure (s) performed / ASA grades.

PASCOM-10 Surgical Treatment Event Count (N) Count (%)
Patients recorded as Discharged 18 72%
Patient not recorded as Discharged 7 28%
Total no. of sample 25 100%
No observed sequella 10 40%
Procedure related complications: (resolved and discharged in all cases) 6 24%
Thickened scar line or painful (SCR) Pain: Scar line hypertrophy / Keloid may not be painful 5 20%
Pain at site of surgery (PNSS): Surgical site beyond six weeks 1 4%
Non-related procedure complications 2 8%
Wound dehiscence recorded for Arthroplasty of 4th distal interphalangeal joint (IPJ) 1 4%
Iatrogenic: Surgery failed e.g recurrence, floating toe, hallux varus recorded for Athroplasty of 2nd proximal IPJ 1 4%
Total no. of patients who had complications 8 25%

Table 3 Surgical Sequelae recorded postoperative.

Any complications recorded at the end of postoperative period on PASCOM-10 is summarised in Table 3. 18 patients (72%) were discharged, of which, 10 (40%) had no postoperative sequelae; 6 (24%) patients had complications relating to the fifth metatarsal scarf osteotomy and; 2 (8%) patients had complications that were not related to the procedure. All 8 (32%) patients who presented with complications have been recorded as discharged. However, clinical decision status for 7 (28%) patients were not recorded thereby, it was unclear if these patients were discharged with or without any complications.

Responsiveness of MOXFQ

As shown on Table 4, the p-values (two-sided) for the W/S, P and SI differences are all <0.001. Therefore, in each domain, the authors have observed a highly significant pre-post difference. The null hypothesis of no effect can thus be rejected in all domains. In the W/S domain, the mean pre-MOXFQ score was 54.7 (SD 21.3), the post-MOXFQ score reduced to 20.2 (SD 27.6). For the P domain, score was reduced from 60.4 (SD 17.9) to 22.2 (SD 21.4) and in the SI category, 50.4 (SD 19.5) to 6.7 (SD 11.2) respectively. A 95% confidence interval for the difference was given by (22.786, 46.254) in W/S; (29.507, 46.893) in P and; (34.534, 52.826) in SI.

Responsiveness of PSQ-10

The mean PSQ-10 score in Table 5 for the cohort was 88.08 with the majority of patients (88%) scoring between 81 and 100 which suggests that good surgical outcomes can be achieved with the fifth metatarsal scarf osteotomy.

The majority of patients (96%) as shown in Table 6 recorded ‘pain relief’ in their response. ‘Improved mobility’ accounted for 16% and ‘better footwear’ 8%. ‘Improved cosmesis’ and ‘Others’ counts occurred less commonly, accounting for 4% respectively.

MOXFQ scores with associated difference scores, 95% confidence interval and p-values.
Domain Pre-mean Pre-SD Post-mean Post-SD Difference Mean Difference SD 95% Confidence interval P-value
W/S 54.68 21.268 20.16 27.559 34.520 28.427 (22.786, 46.254) <0.001
P 60.40 17.907 22.20 21.413 38.200 21.059 (29.507, 46.893) <0.001
SI 50.36 19.506 6.68 11.190 43.680 22.156 (34.534, 52.826) <0.001

Table 4 Summary of MOXFQ scores with associated difference scores, 95% confidence interval and p-values.

Figure 3 Pre/post op comparative MOXFQ distribution. The graph illustrates a significant improvement across the three MOXFQ domains of W/S,P and SI following the procedure. Higher scores in preoperative MOXFQ scores denote greater severity.

Band Count (N) Count (%)
1-10
11-20
21-30
31-40
41-50
51-60 1 4%
61-70
71-80 2 8%
81-90 7 28%
91-100 15 60%
Total sample size 25 100%
Mean PSQ-10 Scores 88.08

Table 5 PSQ-10 Score distribution.

Pain relief Improved mobility Better footwear Improved cosmesis Others
24 (96%) 4 (16%) 2 (8%) 1 (4%) 1 (4%)

Table 6 Total counts for each PSQ010 question 1 response (percentage of all themes).

Question 2-10 n. %
Qn. 2 Patient who stated the risks and possible complications of surgery have been explained to them prior to surgery 25 100%
Qn. 3 Patients who stated they know what to do should a problem arise after postoperatively 25 100%
Qn. 4 Patients who stated they have had problems postoperatively
No 17 68%
Yes, minor 6 24%
Yes, major 2 8%
Qn. 5 Patients who stated some postoperative pain but coped, and those who had minimal or no pain. 25 100%
Qn. 6 Patients who returned to footwear by 2 weeks 5 20%
Patients who returned to footwear by 4 weeks 11 44%
Patients who returned to footwear by 6 weeks 2 8%
Patients who returned to footwear by 8 weeks 4 16%
Patients who returned to footwear by 12 weeks 1 4%
Patients who returned to footwear by 6 months 2 8%
Qn. 7 Patients who described no discomfort or any occasional twinges from their original foot condition 20 80%
Qn. 8 Patients who described their foot condition was better or much better following surgery 24 96%
Patients who stated their foot condition deteriorated or a little worse following surgery 0 0%
Qn. 9 Patients who stated they would have the surgery again under the same conditions 22 88%
Qn. 10 Patients whose expectations were met or partly met 25 100%

Table 7 Summary of answers to key PSQ-10 question 2-10.

Radiographic Analysis

The results of the radiographic analysis for both 5-MTPA and 4-5 IMA are displayed in Table 9. The mean pre 5-MTPA measurement was 19.8 (SD 5.1), the post 5-MTPA measurement reduced to 8.0 (SD 3.1). For the mean 4-5 IMA measurement, it was reduced from 11.6 (SD 1.9) to 5.7 (SD 1.7). Post-operative mean scores of both 5-MTPA and 4-5 IMA fell within the normal angle range for non-pathological foot. A 95% confidence interval for the difference was given by (9.65338, 13.90662) in 5-MTPA and (4.72395, 6.88405) in 4-5 IMA. Improvement of both evaluated angles was highly statistically significant (p-values < 0.001).

Figure 5 Illustrates the significant improvement of the radiographic measurements for both 5- MTPA and 4-5 IMA postoperatively.

XRAY angles Pre-mean Pre-SD Post-mean Post-SD Difference Mean Difference Std.

Deviation

95% Confidence interval P-value
5-MTPA 19.8120 5.05423 8.0320 3.13325 11.78000 5.15194 (9.65338,19.0662) <0.001
4-5 IMA 11.5520 1.94489 5.7480 1.69118 5.80400 2.61653 (4.72395, 6.88405) <0.001

Table 9 Radiological measurement scores with associated difference scores, 95% confidence interval and p-values.

Discussion

The prevalence of tailor’s bunion in the general population is still unknown, although existing literature suggests that the deformity mostly occurs in adults in their 40s and 50s and affects women more than men [35,36]. This is also reflected in the demographic data found in this study (Table 1); a vast majority of patients (84%) are female with an average age of 43.6 y/o (1.dp). Many studies have in fact found women are most likely to suffer with foot pain more than men as not only do they have higher familial tendency to development of structural forefoot deformities, but have also been observed to wear shoes that were too small for their feet [15,18,36]. Increased pressure from shoes over the prominence of the fifth metatarsal head can cause irritation, pain and development of skin lesions such as calluses and corns [35].

The results of the MOXFQ scores in this review showed significant improvement of more than 50% decrease postoperatively with the procedure in all three domains (W/S, P, SI). For the study population, the outcomes obtained demonstrated that the changes for the three MOXFQ domains are beyond the measurement error on a 95% confidence level hence can be interpreted as true changes. Furthermore, the score changes obtained have all exceeded Dawson’s estimated Minimal Clinically Important Differences (MCID) value of W/S, 12 in P and 24 in SI [11]. Scores exceeding the MCID are known to be clinically relevant [11]. This paper also found the procedure clinically effective with high levels of patient satisfaction and improved quality of life as reflected by a mean PSQ-10 score of 88.08 with the majority of patients (88%) scoring between 81 and 100. The high level of subjective satisfaction would be consistent with previous studies which also proved good results with the diaphyseal osteotomy procedure [3,19,22,25,41]. High patient satisfaction was further indicated by results of question 10 of the PSQ-10 where all the patients’ original expectations of surgery had been met or partly met; question 7 where 80% of patients described no discomfort or any occasional twinges from their original foot condition and question 8 where 24 (96%) patients described their foot condition as better or much better.

Patients scored highest for concerns relating to foot pain in comparison to other domains preoperatively on the MOXFQ. This correlates with their response on PSQ-10 question 1 taken at a 6 month postoperative review; where ‘pain relief’ accounted for 24 (96%) of patients’ expectation. This could be due to how services operate in the public sector; in that, cosmetic surgery is not routinely provided on the NHS and, indication for any surgical intervention is only warranted if the pathology presented is symptomatic [31]. While the interpretation of patients’ expectations can be challenging and varies depending on time, health and environmental factors [44]. The PROMs results obtained from this study are similar to other studies in that patients attending for foot surgery generally expect pain relief, followed by improved mobility and shoe fitting [26,44].

Acute postoperative pain is most commonly reported by patients following surgery [23]. Although this could deter patients from undergoing surgery again, 88% of patients from this study (Table 7) stated they would have the surgery again under the same conditions, should the need arise. Furthermore, question 5 of the PSQ-10 shows that 92% of patients reported no or minimal pain, with the remaining 8% reporting having some pain but were able to cope. This suggests that a combination of popliteal nerve block with a three day course of oral analgesia (paracetamol, non-steroidal anti-inflammatory drug (NSAID) and weak opioid) is well tolerated by patients with adequate effects.

From the 25 operated feet, there were no reported intra-operative complications and 6 cases of minor postoperative procedure associated complications. This was recorded on PASCOM-10 as ‘a scar line hypertrophy/keloid which may not be painful’ in 5 cases and pain in the surgical site beyond six weeks in 1 case. These findings now form a part of the consenting process for future patients undergoing this procedure. The two other cases of postoperative sequelae recorded were not a direct complication of the fifth metatarsal scarf, but rather of another procedure performed in the same episode. Overall, this paper recorded low complications which supports findings from systematic and meta-analysis study by Martijn, et al., [29] which reviewed complications arising from proximal, diaphyseal and distal osteotomies for correction of tailor’s bunion and found very low complication rates with diaphyseal osteotomies [29]. Some of the complications recorded on their study included hardware complications (i.e., removal of screws fixation, screw breakage/migrations), painful scar, delayed or non-union, infection and a revision surgery. None of these complications were recorded in this study. Other general postoperative complications such as deep vein thrombosis or pulmonary embolism were also not recorded.

When returning to normal footwear postoperatively, 5 patients returned at 2 weeks, 11 patients at 4 weeks, 2 patients at 6 weeks and 4 patients at 8 weeks, however, 1 patient required 12 weeks and 2 patients required 6 months. The total number of patients who returned by the eighth week was 22 (88%) suggesting the use of two countersunk screws provides sufficient stability thus enabling guarded weightbearing in normal footwear. Although there is currently no research which specifically looked into the mechanical strength of fifth metatarsal scarf osteotomies for correction of tailor’s bunion utilizing one or two screws, the positive results of this study is comparable to all the other studies which utilised screws in their diaphyseal osteotomies [3,19,22,25,41]. The procedure also allows for structural correction of increased 5-MTPA and 4-5 IMA. Radiographic analyses in this study revealed significant improvements in these angles which suggest the procedure has value in obtaining promising correction results in patients with widened 4-5 IMA. Despite the differing weight bearing status between pre- and postoperative films, the radiographic improvement findings are similar to other studies [3,19,22,25,41].

Finally, this review achieved 100% for question 2 of the PSQ-10 which asks patients if the risks and possible complications of surgery were explained to them preoperatively. This strongly indicates that there is a robust consenting process employed by the department – thereby protecting patients from unwanted medical intervention, and also safeguards their rights to autonomy, self-determination and inviolability [20]. This is also reflected on the Standards of Proficiency for podiatrist practicing podiatric surgery section 1.6; which recognizes the importance of delivering clear communication with patients and to ensure that they are fully informed of the proposed treatment benefits, risks and consequences [21]. Especially in the field of surgery, this information may be of relevance to patients’ decision making on the treatment choice and who they wish to seek treatment from [4].

Strengths and Limitations

This service review has a few limitations. It is retrospective in design which sits on the lower levels of evidence hierarchy [32]. It is also limited by a small study sample size as the cohort was determined by timeframes and tailor’s bunions are relatively uncommon compared to other foot and ankle pathologies and, as 28% patients’ final treatment episode on PASCOM-10 were missing thereby, risking a possibility of failing to pick up any complications and concerns. Lastly, the differing weight bearing status of pre- and postoperative films could lead to inaccuracy in radiographic tailor’s bunion angular evaluation following the surgery.

The authors believe that the study has taken a novel approach to evaluating diaphyseal osteotomy procedure by not just utilizing clinical outcomes and radiographic evaluations, but most importantly, has employed both MOXFQ and PSQ-10 functional outcomes instruments known for their sensitivity, reliability and validity when assessing PROMs [12]. In contrast to AOFAS, MOXFQ and PSQ-10 incorporate both subjective and objective components [11]. Furthermore, by using PASCOM-10, the data was easily extracted while keeping patient/service users’ information confidential. PASCOM-10 enables its users to collect anonymous or pseudo anonymous data related to the selected cohort of patients who underwent a specific procedure [7].

Conclusion

This service evaluation demonstrates that the fifth metatarsal scarf osteotomy provides good clinical outcomes, is able to address a range of angular deformity and has low complication rates associated with the procedure. The use of two screws for fixation potentially enhances stability of the osteotomy and allows for early mobilisation following the surgery.

It also highlights the key role of MOXFQ and PSQ-10 PROMs instruments when determining patient satisfaction with the services received alongside the clinical outcomes. It is important to recognise that the isolated use of objective clinical outcomes following surgery can overlook factors which are pertinent to patients and the contribution patients’ perspective can have in healthcare appraisal [12]. Therefore, it is in the interest of the surgical team to know how well they are meeting the needs of their patients in a meaningful way utilizing reliable and valid PROMs instruments to provide them with the information needed to assess the quality and outcome of care.

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Fluoroquinolone-induced Achilles tendinopathy – A case report and management recommendations

by Kaylem M. Feeney1,2 BSc (Hons), MSc, MChSI

The Foot and Ankle Online Journal 13 (4): 12

Fluoroquinolone antibiotics are frequently used in the management of infection despite being associated with several side effects including the potential to cause tendon injury. While numerous case reports of Achilles tendon injury related to fluoroquinolone exposure exist in the literature, there is a paucity of research evaluating the effectiveness of treatment interventions for the condition. The author presents a case of chronic bilateral Achilles tendinopathy associated with two separate exposures to ciprofloxacin and its subsequent management with eccentric loading exercises and extracorporeal shockwave therapy (ESWT).

Keywords: Fluoroquinolone, complication, Achilles, tendinopathy, ESWT

ISSN 1941-6806
doi: 10.3827/faoj.2020.1304.0012

1 – Bon Secours Hospital, Galway, Ireland
2 – School of Medicine, University of Limerick, Ireland
* – Corresponding author: kaylemfeeney1995@gmail.com


Fluoroquinolone antibiotics have long been used in the management of infection due to their broad spectrum of activity [1]. While the fluoroquinolone class of antibiotics are generally well-tolerated, they have been associated with complications including tendon injury [2]. There have been several case reports in the literature of fluoroquinolone-induced Achilles tendinopathy though few have reported on the effectiveness of interventions in this population.

Case Report

A 69-year-old male presented to the outpatient clinic with a 7-year history of pain in both Achilles tendons. The patient was generally fit and healthy and was taking no medication at the time of appointment. The patient gave a background history of a sudden onset of bilateral Achilles tendon pain during hospitalization for sepsis. The patient had been prescribed ciprofloxacin as an inpatient and within 24 hours had extreme pain in both Achilles tendons. Treatment with ciprofloxacin was ceased immediately and the patient commenced on an alternative treatment. Pain in the Achilles tendon decreased significantly over the following 6 months though the patient had a persistent low level pain in both Achilles tendons.

Eight months following the initial onset of Achilles tendon pain, the patient suffered a chest infection. The patient was prescribed ciprofloxacin and approximately 72 hours later had extreme debilitating pain in both Achilles tendons, worse than the initial occasion up to the point where he was unable to walk. Antibiotic therapy with ciprofloxacin was ceased and the patient treated with an alternative antibiotic and the chest infection cleared uneventfully. However, on this occasion, the pain in the Achilles tendons did not improve. The patient was unable to do any significant exercise and had made no significant progress in terms of pain or function over the following 6 years despite rest, stretching, strengthening and physical therapy.

Physical examination showed thickening of the midportion of the Achilles tendon bilaterally with pain on palpation and during a single leg heel raise.

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Figure 1 Sagittal view MRI showing Achilles tendon thickness of 11.8mm.

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Figure 2 Axial view MRI showing Achilles tendon thickness of 11.8mm.

MRI examination showed gross thickening of the Achilles tendon measuring 11.8mm on the sagittal view (Figure 1) consistent with chronic Achilles tendinosis. Axial view MRI also demonstrated significant thickening of the Achilles tendon in keeping with chronic Achilles tendinosis (Figure 2).

On the day of presentation the patient had a VAS score of 7/10 on a daily basis and a Roles and Maudsley score of 4.

The patient was commenced on Alfredson’s protocol [3] for Achilles tendinopathy and treated with three sessions of extracorporeal shockwave therapy (ESWT), which has shown to be effective in the management of non-insertional Achilles tendinopathy [4]. ESWT in this case consisted of treatment with 2,500 impulses at a frequency of 10Hz on three occasions spaced one week apart. At the 3-month follow up the patient reported a reduction in pain levels with a VAS score of 3/10. His Roles and Maudsley score had also improved from a score of 4 to a score of 2. At the 6-month follow up, the patient had a VAS score of 2/10 and a Roles and Maudsley score of 2. While the patient still had pain, this pain had improved significantly suggesting that treatment with eccentric loading exercises following Alfredson’s protocol [3] combined with a course of ESWT may be beneficial in reducing pain and improving function in patients with chronic, recalcitrant fluoroquinolone-induced Achilles tendinopathy.

Discussion

Fluoroquinolone antibiotics are becoming increasingly utilized because of their broad antibacterial spectrum and extensive tissue penetration [5]. The most frequently seen adverse effects include headache, skin reaction and gastrointestinal upset [6]. However, fluoroquinolone use has also been linked to tendon injury and tendon rupture including tendinopathy of the biceps brachii, supraspinatus, extensor pollicis longus and Achilles tendons [7]. The first reported case of fluoroquinolone-associated Achilles tendinopathy was in 1983 [5] and since then more than 100 cases have been reported in the literature [8]. The mechanisms of tenotoxic effects of fluoroquinolone antibiotics are unclear, though three main mechanisms have been proposed in the literature and include tendon ischaemia, degradation of tendon matrix and alteration of tenocyte activity [9]. The cause of Achilles tendinopathy not related to fluoroquinolone use is not fully understood but in the absence of acute rupture in trauma it is generally considered an overuse syndrome due to excessive loading of the tendon during activity [6,8,10,11,12]. Management is primarily conservative with rest, non-steroidal anti-inflammatory medication, steroid injection therapy, stretching, strengthening, prolotherapy, ESWT and platelet-rich plasma injection all being proposed as treatment options in the literature [13,14,15]. Fluoroquinolone-induced Achilles tendinopathy normally resolves within 2 months [15] though prolonged symptoms have been reported in one case lasting two years [15]. The effective management of fluoroquinolone-induced Achilles tendinopathy is not well documented in the literature with the majority of case reports failing to report method of treatment and success rates of treatment [14,15]. Treatment in the literature has focussed on cessation of fluoroquinolone therapy immediately followed by a period of rest and/or immobilisation [15-20]. In this present case, cessation of therapy and subsequent rest was unsuccessful in the improvement of the patient’s symptoms and as such further treatment was required. Alfredson’s protocol [3] has been shown to be effective in the management of non-insertional Achilles tendinopathy [21,22]. The patient also received three sessions of ESWT, spaced one week apart, with 2,500 impulses at a frequency of 10Hz per treatment. The mechanism of action of ESWT and its effect on tendon tissue is not fully understood though there is research to suggest that ESWT promotes neovascularization and has an inhibitory effect on nociceptors in animal models [23,24]. Furthermore, a review of biological studies has suggested that the mechanical stimulation of tenocytes during ESWT stimulates an increase in the production and release of various growth factors and the proliferation in fibroblasts [25]. However, further biological studies are necessary to fully understand the effects of ESWT on human tendon.

In this case, treatment with Alfredson’s protocol [3] combined with a course of ESWT was effective in significantly reducing the patients symptoms over a 3-month period and these improvements were maintained at 6 months. Despite the limitations of a single case study, treatment with a combination of ESWT and eccentric loading following Alfredson’s protocol [3] may be considered in cases where cessation of fluoroquinolone therapy and rest/immobilisation fail to resolve a patient’s symptoms.

Conclusion

Clinicians should be aware of the risk of fluoroquinolone treatment in the development of Achilles tendinopathy. If Achilles tendinopathy is suspected during therapy, cessation of treatment and rest is important in order to prevent progression of tendon damage and worsening of symptoms. If the condition fails to improve, eccentric loading following Alfredson’s protocol combined with a course of ESWT may improve patient symptoms and allow a return to exercise. Further research with a large sample size and longer follow-up is necessary to confirm these promising results.

References

  1. King DE, Malone R, Lilley SH. New Classification and Update on the Quinolone Antibiotic. Am Fam Physician 2000;61(9):2741-2748.
  2. Stahlmann R, Lode H. Toxicity of Quinolones. Drugs 1999; 58(2):37-42.
  3. Alfredson H, Pietilä T, Jonsson P, Lorentzon R. Heavy-Load Eccentric Calf Muscle Training For the Treatment of Chronic Achilles Tendinosis. Am J Sports Med. 1998;26(3):360-366.
  4. Rasmussen S, Christensen M, Mathiesen I, Simonson O. Shockwave Therapy for Chronic Achilles Tendinopathy: A Double-Blind, Randomized Clinical Trial of Efficacy. Acta Orthop 2008;79(2):249-256.
  5. Bailey RR, Kirk JA, Peddie BA. Norfloxacin Induced Rheumatoid Disease. N Z Med J 1983;96(736):590.
  6. Yu C, Giuffre BM. Achilles Tendinopathy After Treatment With Fluoroquinolone. Australasian Radiology 2005;49(5):407-410.
  7. Van Der Linden PD, Van Puijenbroek EP, Feenstra J. Tendon Disorders Attributed to Fluoroquinolones: A Study on 42 Spontaneous Reports in the Period 1988-1998. Arthritis Care Res 2001;45(3):235-239.
  8. Tam PK, Ho CTK. Fluoroquinolone-Induced Achilles Tendinitis. Hong Kong Med J 2014; 20(6):545-547.
  9. Childs SG. Pathogenesis of Tendon Rupture Secondary to Fluoroquinolone Therapy. Orthop Nurs 2007;26(3):175-182.
  10. Kim GK, Del Rosson JQ. The Risk of Fluroquinolone-Induced Tendinopathy and Tendon Rupture: What Does the Clinician Need to Know? J Clin Aesthet Dermatol 2010;3(4):49-54.
  11. Mafulli N, Sharma P, Luscombe KL. Achilles tendinopathy: Aetiology and Management. J R Soc Med 2004;97(10):472-476.
  12. Horn A, McCollum G. Achilles Tendinopathy – Part 1: Aetiology, Diagnosis and Non-Surgical Management. SA Orthop J 2015;14(3):24-31.
  13. Roche AJ, Calder JDF. Achilles Tendinopathy: A Review of the Current Concepts of Treatment. J Bone Joint Surg 2013;95(10):1299-1307.
  14. Lewis T, Cook J. Fluoroquinolones and Tendinopathy: A Guide for Athletes and Sports Clinicians and a Systematic Review of the Literature. J Athl Train 2014; 49(3):422-427.
  15. Khaliq Y, Zhanel GG. Fluroquinolone-Associated Tendinopathy: A Critical Review of the Literature. Clin Infect Dis 2003;36(11):1404-1410.
  16. Zabraniecki L, Negrier I, Vergne P, Arnaud M, Bonnet C, Bertin P, Treves R. Fluoroquinolone Induced Tendinopathy: Report of 6 Cases. J Rheumatol, 1996;23(3):516-520.
  17. Ribard P, Audisio F, Kahn MF, De Bandt M, Jorgensen C, Hayem G, Meyer O, Palazzo E. Seven Achilles Tendinitis Including 3 Complicated by Rupture During Fluroquinolone Therapy. J Rheumatol 1992;19(9):1479-1481.
  18. Meyboom RHB, Olsson S, Knol A, Dekens-Konter JAM, De Koning GHP. Achilles Tendinitis Induced by Pefloxacin and other Fluoroquinolone Derivatives. Pharmacoepidemiol Drug Saf 1994;3:185-189.
  19. Carrasco JM, Garcia B, Andujar C, Garrote F, de Juana P, Bermejo T. Tendonitis Associated with Ciprofloxacin. Ann Pharmacother 1997;31(1):120.
  20. Lewis JR, Gums JG, Dickensheets DL. Levofloxacin-Induced Bilateral Achilles Tendonitis. Ann Pharmacother 1999;33(7):792-795.
  21. Stevens M, Tan CW. Effectiveness of the Alfredson Protocol Compared with Lower Repetition-Volume Protocol for Midportion Achilles Tendinopathy: A Randomized Controlled Trial. J Orthop Sports Phys Ther 2014;44(2):59-67.
  22. Van der Plas A, de Jonge S, de Vos RJ, van der Heide HJL, Verhaar JAN, Weir A, Tol JL. A 5-year Follow-Up Study of Alfredson’s Heel-Drop Exercise Programme in Chronic Midportion Achilles Tendinopathy. Br J Sports Med 2012;46(3):214-218.
  23. Wang CJ, Huang HY, Pai CH. Shock Wave Enhances Neovascularization at the Tendon-Bone Junction. J Foot Ankle Surg 2002;41(1):16-22.
  24. Wang CJ, Wang FS, Yang KD, Huang CS, Hsu CC, Yang LC. Shock Wave Therapy Induced Neovascularization at the tendon-Bone Junction: A Study in Rabbits. J Orthop Res 2003;21(6):984-989.
  25. Notarnicola A, Moretti B. The Biological Effects of Extracorporeal Shock Wave Therapy (ESWT) on Tendon Tissue. Muscles Ligaments Tendons J 2012;2(1):33-37.

 

Reconstruction of an Achilles rupture with 12 cm defect utilizing Achilles tendon allograft and calcaneal bone block: A case report

by Isaiah Song1*, DPM; Alvin Ngan2, DPM, FACFAS

The Foot and Ankle Online Journal 13 (4): 11

Chronic Achilles tendon ruptures, especially with extensive defects, are challenging to repair, and options are limited. We present a case of a neglected Achilles tendon rupture with a 12 cm defect, treated with an Achilles tendon allograft with a calcaneal bone block. The repair was augmented with a flexor hallucis longus (FHL) tendon transfer as well as human acellular dermal matrix. At 1-year follow-up the patient had no pain and was able to walk 2 miles at a time. There was no re-rupture of the affected limb, infection or allograft morbidity.

Keywords: Achilles tendon, chronic, tendon allograft, tendon rupture, surgical technique, FHL tendon transfer

ISSN 1941-6806
doi: 10.3827/faoj.2020.1304.0011

1 – Resident, Swedish Foot & Ankle Residency Program, Swedish Medical Center, Seattle, WA
2 – Attending Physician, Swedish Foot & Ankle Residency Program, Swedish Medical Center, Seattle, WA
* – Corresponding author: isaiahsong@gmail.com


A chronically ruptured or neglected Achilles tendon is defined as a rupture with 4-6 weeks between the time of injury and treatment [1,2]. An estimated 20-35% of Achilles ruptures have a delayed diagnosis due to unrecognized injury, misdiagnosis or late presentation [1,2]. Between injury and treatment, granulation tissue between tendon ends prevents apposition and fibrous tissue develops in the rupture site [3-5]. The triceps surae muscle continues to contract and the proximal tendon stump retracts and adheres to the surrounding fascia [6, 7]. Unrecognized injury and retraction of the tendon stump may result in large defects.

Various techniques have been described for surgical repair of the neglected Achilles tendon rupture including gastrocnemius tendon advancement, turndown flaps, autografts, allografts and tendon transfers [8-12]. Which technique provides the best outcome is unknown, and some techniques are limited to smaller defects.

We present a case of a chronic Achilles rupture with a 12 cm defect reconstructed with an Achilles tendon allograft with calcaneal bone block, augmented with a flexor hallucis longus (FHL) tendon transfer and human acellular dermal matrix.

Case Study

A healthy, very active, 71-year-old male initially presented with ankle weakness and difficulty with gait. He was treated by an outside provider for one year with presumed Achilles tendonitis. However, at presentation at the current attendings clinic, he had a palpable defect with a positive Thompson’s test for an Achilles rupture. There was minimal calf atrophy compared to the contralateral side. His gait was antalgic and apropulsive with poor balance. MRI demonstrated an Achilles rupture with approximately 9 cm retraction. The patient was initially offered permanent bracing because of his age. However, due to his good health, and very active lifestyle, he elected for surgical repair understanding the potential limitations of achieving a full recovery given the longstanding misdiagnosis. The patient was counseled on the probable use of allograft and tendon transfer because of the extensive defect.

Figure 1 Intraoperative appearance of the chronic rupture site, interposed with fatty and mucoid diseased tissue or “pseudotendon”.

Figure 2 The proximal excised portion of the diseased Achilles.

Surgical technique

The patient was placed prone on the operative table under general anesthesia with a thigh tourniquet. A linear incision was made just medial to the midline of the Achilles tendon and deepened to expose the tendon. The rupture site was identified. The defect had filled with interposed scar tissue, with fibrofatty and mucoid consistency. This non-viable tissue was excised proximally to the level of the gastrocnemius aponeurosis which was noted to be healthy. The degenerated tissue extended distally to the Achilles insertion, with minimal healthy tendon attachment to the calcaneus. Following debridement and excision of diseased tissue, the defect measured 12 cm with the ankle in near maximal plantarflexion.

Figure 3 Additional excision of nonviable, calcified degenerated tissue at the distal Achilles stump.

A frozen Achilles tendon allograft with calcaneal bone block was thawed and pre-tensioned to minimize viscoelastic creep. The calcaneal block portion of the allograft was fixated first, by creating a rectangular cut-out for insertion in the superior aspect of the patient’s calcaneus. The calcaneal bone block was tamped into place and fixated with 2 crossed 3.5 cortical screws with washers.

Before attachment of the allograft proximally, the ankle was placed in 30 degrees of plantarflexion to create a tensioned repair. The proximal portion of the Achilles allograft was then sutured to the gastrocnemius aponeurosis with approximately 3 inches of overlap utilizing a combination of simple interrupted and Krackow suturing, with #2, and #2-0 fiberwire.

In order to improve strength and vascularity to the repair, an FHL transfer was also performed. The FHL tendon was harvested by releasing at the level of the posterior talus, sewn alongside the medial aspect of the Achilles at anatomic tension. The musculotendinous portion of the FHL was also sutured to the undersurface of the proximal repair site with the intent to bring vascularity closer to the repair.

Finally, a human acellular dermal matrix product was sewn over the proximal repair with 3-0 Vicryl, to provide reinforcement, and scaffolding for host tissue ingrowth. The incision was irrigated, the tourniquet was released and the patient was placed into a non-weight bearing compression splint with anterior and posterior slabs.

Figure 4 Planning of graft placement following excision of the chronic rupture.

Figure 5 The proximal Achilles allograft was sutured into the gastrocnemius aponeurosis after securing the distal calcaneal block with 2 crossed screws.

Figure 6 A human cellular dermal matrix was overlaid as the final step.

Figure 7 Patient demonstrating ability to perform partial heel rise on the reconstructed side.

Figure 8 Patient demonstrating ability to perform heel rise on the contralateral side for comparison.

Post-operative protocol

The patient was splinted for 14 days and remained non-weight bearing. Following suture removal, the patient was casted in plantarflexion, remaining non-weight bearing for the next 4 weeks. At 6 weeks, the patient was referred to physical therapy and was also transitioned to weight bearing in a walking boot with heel lifts and progressed gradually to neutral by decreasing the heel lifts. At 12 weeks, he was transitioned out of a boot to a shoe.

Results

The patient had no complications during follow-up. At 1 year follow-up, the patient reported no pain and was able to return to normal daily activities, and walk 2 miles. His range of motion was symmetrical to the contralateral side, and manual muscle testing revealed only slight weakness. He was able to perform heel rise symmetrical to his contralateral side. X-rays showed incorporation of the calcaneal block graft. He was overall pleased with the surgery.

Figure 9 Postoperative x-ray showing incorporation of the calcaneal bone block portion of allograft.

Interestingly, he was more bothered by contralateral Achilles pain. Initially this was presumed to be compensatory tendonitis. After failing conservative care, he had ultrasound evaluation, as MRI was contraindicated due to interval placement of a pacemaker. His contralateral Achilles showed findings consistent with tendinosis or chronic tearing. During this ultrasound, his reconstructed Achilles was also examined and it showed expected findings of stable appearing heterogeneous texture of the Achilles allograft with no rupture.

While the patient was satisfied with his reconstructed Achilles, he felt he was more limited by his contralateral Achilles tendinosis and eventually elected for surgery on that side as well.

Discussion

Large defects following Achilles ruptures are challenging. Delay in diagnosis may lead to retraction of the tendon stump, and atrophy of the gastrocnemius muscle. Furthermore, if significant tendinosis was present prior to rupture, the actual defect may be larger than presumed following debridement. Ofili in 2016 reported that MRI underestimates the true extent of Achilles tendinosis [18]. Indeed, our patient had a 12 cm defect following debridement despite MRI initially predicting a 9cm defect.

While smaller defects may be typically treated by gastrocnemius advancement or flap, there is no consensus on how to manage larger Achilles defects. In the author’s experience, gastrocnemius advancement techniques allow repair of only up to 6-8 cm defects. Our patient had a 12 cm defect and therefore, with limited repair options, it was felt appropriate to utilize Achilles tendon allograft. Additionally, given the significant disease in the distal stump at the insertion, there was no viable tissue to suture the Achilles allograft, and therefore the calcaneal bone block proved useful for distal reattachment. One risk of a calcaneal bone block would be delayed or non-union. Deese in 2015 and Ofili in 2016 have reported delayed union with incorporation at the calcaneus. [12,19] Our patient showed radiographic healing at 6 weeks post-op.

Reconstruction using an Achilles tendon allograft with a calcaneal bone block has previously been demonstrated to have good results [18,19]. These studies did not include an FHL transfer. The FHL transfer is a relatively simple, in-phase transfer with the potential benefits of increasing strength of the repair and providing additional plantarflexion power. Additionally, the FHL transfer theoretically may provide additional vascularity from the flexor hallucis muscle belly to the repaired Achilles. The FHL transfer has been shown to have high patient satisfaction and minimal donor morbidity has been noted with this procedure [21-24]. From a technical standpoint, the use of a calcaneal bone block with screw fixation may limit the ability to secure an FHL transfer with biotenodesis as creating an additional bone tunnel adjacent to the screws may create stress risers. Therefore in the current case the FHL tendon was sutured side by side instead.

Disadvantages with allograft procedures are the risk of disease transmission, longer allograft incorporation time, and increased cost. There is also a potential amount of creep in allograft tendons. In addition, Hanna in 2014 reported that 4 of 6 patients with an Achilles allograft with calcaneal bone block ambulated with a limp and complained of weakness at 16-32 months [18]. It is important to counsel patients with longstanding neglected ruptures, that recovery of full strength may not be possible. Our patient, however, appeared to be able to perform a symmetrical appearing single heel rise on examination.

In our case, we augmented the allograft repair with FHL transfer and human acellular dermal matrix. Human acellular dermal matrix acts as a scaffold for host revascularization and cellular growth. [25] A few studies have described acellular dermal matrix augmentation to strengthen an Achilles tendon rupture site, all with favorable outcomes without any re-ruptures. [26-29] Therefore, the final addition of the dermal matrix in our patient was intended to assist with incorporation of the large allograft.

In conclusion, Achilles tendon reconstruction with tendon-bone block allograft augmented with FHL transfer and a human acellular dermal matrix may successfully repair a severely degenerated and neglected Achilles tendon rupture. We believe this technique can be useful for Achilles tendon ruptures with large deficits up to, and perhaps more than 12 cm.

Acknowledgements

Thank you to the Swedish Medical Center Foot and Ankle Surgery Residency Program for the support. Additional thanks to Drs. Brian Rougeux and Bryn Rowe for early input into this case study.

References

  1. Maffulli N. Clinical tests in sports medicine: more on Achilles tendon. Br J Sports Med. 1996;30(3):250.
  2. Maffulli N, Ajis A. Management of chronic ruptures of the Achilles tendon. J Bone Joint Surg Am. 2008;90(6):1348–1360.
  3. Carden DG, Noble J, Chalmers J, Lunn P, Ellis J. Rupture of the calcaneal tendon. The early and late management. J Bone Joint Surg Br. 1987;69(3):416–420.
  4. Bosworth DM. Repair of defects in the tendo achillis. J Bone Joint Surg Am. 1956;38-A(1):111–114.
  5. Yasuda T, Kinoshita M, Okuda R. Reconstruction of chronic Achilles tendon rupture with the use of interposed tissue between the stumps. Am J Sports Med. 2007;35(4):582–588.
  6. Abraham E, Pankovich AM. Neglected rupture of the Achilles tendon. Treatment by V-Y tendinous flap. J Bone Joint Surg Am. 1975;57(2):253–255.
  7. Zadek I. Repair of old rupture of the tendo Achilles by means of fascia lata: Report of a case. JBJS. 1940;22(4):1070.
  8. Maffulli N, Longo UG, Maffulli GD, Rabitti C, Khanna A, Denaro V. Marked pathological changes proximal and distal to the site of rupture in acute Achilles tendon, Knee Surg Sports Traumatol Arthrosc 19, 680–687 (2011). https://doi.org/10.1007/s00167-010-1193-2
  9. Kader D, Saxena A, Movin T, Maffulli N. Achilles tendinopathy: some aspects of basic science and clinical management. Br J Sports Med. 2002;36(4):239–249.
  10. Kannus P, Józsa L. Histopathological changes preceding spontaneous rupture of a tendon. A controlled study of 891 patients. J Bone Joint Surg Am. 1991;73(10):1507–1525.
  11. Aström M, Rausing A. Chronic Achilles tendinopathy. A survey of surgical and histopathologic findings. Clin Orthop Relat Res. 1995;(316):151–164.
  12. Ofili KP, Pollard JD, Schuberth JM. The neglected Achilles tendon rupture repaired with allograft: A review of 14 cases. J Foot Ankle Surg. 2016;55(6):1245–1248.
  13. Duhamel P, Mathieu L, Brachet M, Compere S, Rigal S, Bey E. Reconstruction of the Achilles tendon with a composite anterolateral thigh free flap with vascularized fascia lata: a case report. J Bone Joint Surg Am. 2010;92(15):2598–2603.
  14. Nellas ZJ, Loder BG, Wertheimer SJ. Reconstruction of an Achilles tendon defect utilizing an Achilles tendon allograft. J Foot Ankle Surg. 1996;35(2):144–148; discussion 190.
  15. Lepow GM, Green JB. Reconstruction of a neglected Achilles tendon rupture with an Achilles tendon allograft: A case report. J Foot Ankle Surg. 2006;45(5):351–355.
  16. Hansen U, Moniz M, Zubak J, Zambrano J, Bear R. Achilles tendon reconstruction after sural fasciocutaneous flap using Achilles tendon allograft with attached calcaneal bone block. J Foot Ankle Surg. 2010;49(1):86.e5-10.
  17. Beals TC, Severson EP, Kinikini D, Aoki S. Complex Achilles reconstruction for massive soft tissue loss: allograft, autograft, and use of a temporary cement spacer. J Orthop Trauma. 2010;24(8):e78-80.
  18. Hanna T, Dripchak P, Childress T. Chronic Achilles rupture repair by allograft with bone block fixation: technique tip. Foot Ankle Int. 2014;35(2):168–174.
  19. Deese JM, Gratto-Cox G, Clements FD, Brown K. Achilles allograft reconstruction for chronic Achilles tendinopathy. J Surg Orthop Adv. 2015;24(1):75–78.
  20. Den Hartog BD. Flexor hallucis longus transfer for chronic Achilles tendonosis. Foot Ankle Int. 2003;24(3):233–237.
  21. Hahn F, Maiwald C, Horstmann T, Vienne P. Changes in plantar pressure distribution after Achilles tendon augmentation with flexor hallucis longus transfer. Clin Biomech (Bristol, Avon). 2008;23(1):109–116.
  22. Coull R, Flavin R, Stephens MM. Flexor hallucis longus tendon transfer: evaluation of postoperative morbidity. Foot Ankle Int. 2003;24(12):931–934.
  23. Richardson DR, Willers J, Cohen BE, Davis WH, Jones CP, Anderson RB. Evaluation of the hallux morbidity of single-incision flexor hallucis longus tendon transfer. Foot Ankle Int. 2009;30(7):627–630.
  24. Steginsky BD, Van Dyke B, Berlet GC. The missed Achilles tear: Now what? Foot Ankle Clin. 2017;22(4):715–734.
  25. Norton LW, Babensee JE. Innate and adaptive immune responses in tissue engineering. In: Fundamentals of Tissue Engineering and Regenerative Medicine, pp. 721–745, edited by U Meyer, T Meyer, J Handschel, HP Wiesmann, Springer-Verlag, New York, 2009.
  26. Lee DK. Achilles tendon repair with acellular tissue graft augmentation in neglected ruptures. J Foot Ankle Surg 2007;46:451–455.
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  29. Huang X, Huang G, Ji Y, Ao R, Yu B, Zhu YL. Augmented repair of acute Achilles tendon rupture using an allograft tendon weaving technique. J Foot Ankle Surg 2015;54:1004–1009.

 

Clubfoot in children: An overview

by Mohammad A. Hegazy1, Hossam M. Khairy1, Abdelmonem A. Hegazy2*, Sherif M. El-Aidy1

The Foot and Ankle Online Journal 13 (4): 10

Clubfoot is a case of complex defects affecting mostly newborns and children. Its management represents a great challenge especially in cases of severe resistant cases. Unfortunately, it did not receive the necessary attention in text-books and literature regarding its all aspects under one title. In this article, we aimed to highlight clubfoot focusing pathological anatomy of clubfoot and consequent management options. This might help physicians and surgeons in proper management. Clubfoot shows main anatomical defects including foot cavus, adduction, varus and equinus; the most prominent bony defect has been found in foot talus. Most cases respond to conservative reconstruction including the Ponseti method. However, some cases including those associated with other congenital anomalies are severe and resistant to such conservative management. These cases are suggested to be managed by talectomy that represents a salvage procedure to give a plantigrade foot.

Keywords: clubfoot, anatomy, pathology, talectomy

ISSN 1941-6806
doi: 10.3827/faoj.2020.1304.0010

1 – Orthopedic Surgery Department, Faculty of Medicine, Zagazig University, Zagazig, Egypt.
2 – Anatomy and Embryology Department, Faculty of Medicine, Zagazig University, Egypt.
* – Corresponding author: dr.abdelmonemhegazy@yahoo.com


Clubfoot called talipes equinovarus represents a global health problem affecting 1-2/1000 of live births all over the world. It involves both feet in about half of the reported cases. In unilateral cases, the club foot slightly more affects the right side than the left. It also affects males more than females. The case is mostly managed through conservative measures including the Ponseti method [1]. However, such conservative methods are often unsuccessful for correction of deformity in cases of severe rigid clubfoot [2].

Because of unresponsiveness to many options of management, cases of severe rigid equinovarus deformity represent a challenging problem for orthopedic surgeons. Understanding the pathological anatomy of such cases facilitates their management. There are many surgical options for correction of the deformity including release of soft tissues, Ilizarov correction using an external fixator and triple arthrodesis [3]. However, all these measures also fail to obtain a stable plantigrade foot. Therefore, talectomy has been adopted as a salvage procedure aiming to correct or minimize the deformity. This procedure was used for treatment of deformities of paralytic calcaneovalgus [4].

Although many researchers have tried to clarify the pathogenesis of congenital clubfoot, the exact cause is still obscure. Most of cases (about 80%) occur in normal physical and mental children [5].

In this review, we highlight the clubfoot and its pathological anatomy in a trial for more understanding of its pathogenesis, and hence its proper management. In addition, the options for management were reviewed with a focus on the role of talectomy in severe and resistant cases.

Methods

The clubfoot was investigated through the database of PubMed, Google Scholar, Web of Science, Scopus and others. Data were collected, represented and discussed into the following main subtitles: epidemiology, etiology, pathological anatomy, diagnosis, classification, features and management.

Results and Discussion

Epidemiology of Clubfoot

Clubfoot is one of the commonest and oldest recognized orthopedic anomalies. It was recognized as early as the ancient Egyptians. It has been reported that Egyptian Pharaohs Siptah and Tutankhamun were suffering from clubfoot. The first medical document describing the case was produced in 400 BC by Hippocrates [6].

Clubfoot is encountered in about one per thousand of the live births, varying from one race to another. It affects approximately 150,000-200,000 of newborns all over the world each year. About 80% of cases are encountered in developing countries. It affects both limbs in about 50% of cases. The unilateral clubfoot occurs in the right lower limb more than the left one with a ratio of 2-1 respectively [7]. It might be isolated congenital defect or associated with other serious anomalies especially if it is severe and bilateral [8].

Moreover, the incidence is more in males than females with a ratio of 2-1 respectively. The prevalence in Western Europe and the USA accounts 1-1.4/1000 live births. A lower incidence has been detected in Chinese and Japanese but a higher amongst Polynesian as well as South Africa black populations [5].

Etiology of clubfoot

Clubfoot is classified into two main types; congenital and acquired. Acquired form isn’t inborn-error. It might be caused by associated diseases. These include vascular causes such as Volkmann Ischemic Paralysis and neurogenic diseases comprising poliomyelitis, meningitis, sciatic nerve damage. Congenital clubfoot could be subdivided according to their causes into idiopathic or non-idiopathic types. Idiopathic clubfoot is mostly an isolated birth defect. The causes of non-idiopathic clubfoot include teratologic anomalies, generalized syndromes (e.g. diastrophic syndrome) and neurological diseases of known defects (such as spina bifida). The cases of non-idiopathic clubfoot are commonly associated with the presence of other anomalies with poor response to management either conservative or operative treatment [9].

Many theories have been proposed to explain causes of idiopathic type of clubfoot occurring in normal newborns. One of these theories is the mechanical one laid by Hippocrates which assumes that clubfoot might be caused by an increased intrauterine pressure during pregnancy [10]. This theory is disputed because of absence of association of clubfoot with most cases of overcrowded uterus such as cases of twins, large babies or polyhydramnios [9].

Smoking of mothers at pregnancy might be a cause of increased risk of clubfoot [11]. Another cause might be presence of an aberrant muscle, noticed at surgery to be inserted into the deep fascia of foot opposite the medial side of calcaneus [12]. Zimny, et al., found abnormal contracted plantar fascia with fibroblastic contracture similar to that found in Dupuytren’s disease [13].

There are some findings supporting the genetic factor in etiology of clubfoot. These observations include the increased incidence in cases of previous family history. Such history was found in about 25% of isolated cases of clubfoot. Moreover, there is a coincidence of clubfoot of monozygotic twins of about 33% compared with only 3% in dizygotic. It has been suggested that a variety of apoptotic genes are involved in cell death cascade and consequent shaping the defects in clubfoot [14].

Another study attributed the idiopathic clubfoot to be due a disturbance in the germ cells causing arrest of the foot development at the 5-week stage of fetal life. At this period, called physiological clubfoot stage, the foot bones resemble the shape and position of clubfoot [15]. Similarly, Victoria-Diaz and Victoria-Diaz, stated that the development of the human foot passes into three stages in-utero [16]. In the first (the 15-mm embryo length) stage, the foot appears in the same line with the leg. In the second “embryonic” stage (30-mm embryo length), the lateral side of the leg elongates more in relation to the medial aspect causing the foot to assume the clubfoot position. By the third “fetal” stage (50-mm), the medial side of the leg and foot develops to correct the position assuming that seen normally in the newborn.

Arthrogryposis

It is a common cause for rigid talipes equinovarus found since birth. Its disorders include multiple joints so is commonly called arthrogryposis multiplex congenita. It affects two or more joints with no obvious cause or pathogenesis. There is no specific diagnosis, but this depends mainly on clinical findings [17]. The affected joints involve the large joints of limbs such as hip, knee, ankle, shoulder, elbow and wrist as well as the joints of the foot and hand. The joints of the ankle and foot are mostly affected leading to deformity of clubfoot. Such deformity is resistant to manipulation and conservative measures. It could be improved by radical soft tissue surgery; however, the relapses’ rate is high. Therefore, talectomy is recommended as a salvage or even primary procedure in such cases of severe rigid clubfoot [18].

Spina bifida

Spina bifida is a congenital anomaly in which there is failure of fusion of the two halves of the neural arch of one or more vertebrae. It includes many types [Figure 1]. Spina bifida occulta is a non-symptomatic condition and was discovered at investigation. Other types include meningocele with protrusion of sac of meninges through the vertebral defect and meningomyelocele comprising the nervous tissue and meninges’ protrusion [19]. It represents one of the non-idiopathic causes of rigid and resistant clubfoot. This foot deformity occurs in about 30-50% of cases of spina bifida. It is evident at birth; and its incidence differs according to the site of spina bifida lesion. Clubfoot is noticed in about 90% of cases in case thoracic and lumbar spina bifida whilst the sacral region is associated with clubfoot in 50% of patients.

Figure 1 Types of spina bifida.

Figure 2 Talo-calcaneal (TC) and talo-first metatarsal (TF) angles: A) Normal foot; B) Clubfoot.

Since the case is rigid, non-surgical management including stretching, splinting and serial casting commonly fails. It might be an indication for surgical interference [20].

Pathological anatomy of clubfoot

Despite the cause of the clubfoot is still uncertain to treat the cases accordingly, knowledge of the pathological anatomy remains the main guide in repair and management. Many theories were proposed to explain the bony changes occurring in the foot.

Scarpa [21] mentioned that the osseous defects in the talus are the primary causative factor in pathogenesis of the clubfoot. This suggestion has been supported by other studies later on. They added that talus defects are noticed in all clubfoot types [15].

Other authors postulated that the calcaneus is the primary fault in pathogenesis of clubfoot attributing their suggestion to the ossification of the calcaneus that noticed to appear before that of talus [22].

In clubfoot, there is a complex musculoskeletal alteration in the foot to be directed down and medially resembling the club used to hit the golf ball. The anatomical deviations are summarized by the 4 letters of the word “cave” (C=Cavus, A=Adduction, V=Varus and E=Equinus) (Figure 2) [8].

The main features include the followings (Figures 2,3):

1- Cavus: It is the increased convexity (or longitudinal arch) of the foot. This is caused by increased plantar flexion of the first metatarsal bone in relation to the hindfoot. It is caused by contracture of the plantar aponeurosis. Also, there is contracture of the plantar and spring ligaments.

2- Adduction: The forefoot is adducted. The cuneiforms and metatarsals are deviated towards the midline but they appear of normal shape.

3- Varus: It is the inversion and adduction of the hindfoot. In other words, the heel forms varus angulation. The calcaneus is adducted, plantar flexed and rotated inwards below the talus to lie nearly in the same line.

4- Equinus: The entire foot shows an increased plantar flexion at the ankle joint.

The soft structures on the medial and posterior aspects of the foot are shortened and thickened keeping the position of the foot in adduction and varus with equinus respectively. These structures include deltoid, talonavicular and spring ligaments as well as tibialis posterior tendon medially. Also, there is tightness of the posterior gastrocsoleus complex [8].

Joints of the foot are distorted due to malposition of its bones. The equinus deformity occurs mainly at the ankle joint, but others also particularly subtalar joints contribute to the deformity of the foot. The talo-calcaneo-navicular joint is dislocated with contracture of the soft tissue surrounding it with the ankle. Such contracture includes the joint capsule, ligaments, tendons and their sheaths and muscles. The contractures are noticed in talofibular, calcaneofibular, spring, deltoid and plantar bifurcated Y ligaments as well as the tendo Achilles. Moreover, there are plantar contractures affecting the intrinsic flexors of the toes, abductor hallucis and plantar aponeurosis [9].

Figure 3 Normal foot compared with varus, cavus and equinus.

Figure 4 Diagrams showing superior view of talus in normal and clubfoot.

Talus

Rehman and Faruqui found that all clubfeet show nearly the same defects of foot skeleton. The most prominent features are the small sized foot and distorted talus [23]). The talus is distorted in size, shape and orientation in case of clubfoot. Its head and neck are smaller than normal and directed down and medially. Talus is directed in a plantar flexion position. The trochlear articular surface of the body of the talus is less convex. Despite any change in the shape of the talus body, it is reduced in size [22].

The most apparent change in the talus is found in its anterior part (Figure 4). The neck of the talus is always short, directed medially and plantarward on the body. It is sometimes not apparent. The angle between the long axis of the neck and that of the body is much reduced; in clubfoot it is about 115-135° versus normal foot of 150-155°. The articular surface for navicular is no longer directed forwards like that of normal foot, but deviated medially and plantarward [23]. In case of clubfoot, the degree of medial deviation of the head and neck of the talus is more significant than that of the plantar one. Moreover, the volume of talus in congenital clubfoot is much reduced than that of the normal foot [24].

The navicular bone is dislocated medially lying opposite the tibial malleolus. This causes the front of the head of the talus to be uncovered and pointed towards the lateral instead of medial side [5].

Cuboid is also displaced medially along with the anterior end of the calcaneus displaying foot lateral convexity [9].

Calcaneus

The calcaneus in clubfoot is generally of normal shape but slightly smaller than that of normal foot. It is shifted into equinus, varus and medial rotation along the distorted neck of talus [23]. Epeldegui also noticed a small-sized calcaneus; but added the presence of another deformity in the calcaneus represented by twisting along its long axis with consequent rotational deformities of foot longitudinal axis [25].

Diagnosis of Clubfoot

Prenatal diagnosis

Although most cases of clubfoot are diagnosed at birth, they could nowadays be recognized prenatally. Ultrasonography (US) advent into health care enables the physicians to recognize the case during the intrauterine life. Prenatal US examination has a positive predictive value over 80% with no false results [8]. However, using US at pregnancy doesn’t differentiate between its grades. Its importance lies in preparing the parents to be ready to the degree of postnatal management as early as possible. Also, discovery of the case in-utero motivates the doctors to search about the other congenital anomalies that might be associated with clubfoot in up to 50% of affected fetuses [26]. Diagnosis of the case prenatally could be recognized around the twenty-two weeks of gestation. Once noticed, assurance of the parents is essential, focusing that the case is treatable and not an indication for termination of pregnancy. The parents should know that the clubfoot is not a disabling condition but requires patience, compliance and frequent visits and follow-up to achieve excellent results. Prenatal counseling also is important to offer for the parents the options of treatment and results expected from each line management [27].

Postnatal clinical examination

Immediately after birth, the typical clubfoot is diagnosed by the orthopedic physician through taking full history from the parents and inspection of the shape of the foot. Then, the doctor does palpation of foot bones and surrounding connective tissues for abnormal position and contractures respectively. The affected cases should be thoroughly investigated from the head to the foot toes to exclude the other associated congenital defects. All body joints are examined for presence of contracture characterizing arthrogryposis [7]. The case also should be differentiated from paralytic clubfoot such as multiple congenital malformations. The main aim of assessment of cases is to differentiate the postural talipes from the true clubfoot and to define its severity. Postural type is usually easily correctable to the normal anatomical state at birth or in the infantile period after manipulative strapping [28]. Assessment of movements of foot such as inversion/eversion, adduction/abduction of the forefoot, supination/pronation is important. Evaluations of the gait in neglected cases, range of movements and static weight bearing alignment as well as noticing the differences in-between the two limbs are also valuable [29].

Radiological assessment

Though many radiological modalities have been introduced in health investigations, the clinical assessment remains the more informative one in cases of clubfoot. Up till now, there is no consensus regarding the great value of x-ray in routine evaluation and management of such cases. The standard radiograph does not give an accurate single method for evaluation and further management of cases of clubfoot [30]. This is because most tarsal bones are not ossified at birth except the talus and calcaneus of which ossified centers appear in the plain radiographs as rounded ossicles. However, the ossification centers of metatarsal bones are evident at birth; and become sufficiently ossified by the age of 3 to 4 months [31].

The information achieved from radiological examination is usually taken from standing anteroposterior and lateral views. Specific measurements are taken for assessment of cases of clubfoot. These include the angle between talus and calcaneus in lateral and anteroposterior planes as well as the relation of equinus of calcaneus to the longitudinal axis of tibia. The angle between the long axis of first metatarsal and that of talus (called Meary’s angle) is an indication for cavus for forefoot [29].

Magnetic resonance imaging (MRI) and ultrasound also have been used in evaluation of management. MRI is introduced particularly to detect the gradual response for conservative treatment. However, it is important to remember that the initial decision of treatment depends mainly on the clinical grounds [8].

It is important to note that even in well-corrected cases, some radiological residual defects might persist for a long time in the well-treated cases. These include a small volume of tarsal bones and flattening of talar dome [8].

Classification and Features of Clubfoot

Although there is no agreement about the methods of scoring or classification of clubfoot till now, it’s essential to adopt one of them to predict the appropriate line of management and to assess the progress of the lesion and its prognosis [31].

There are many methods of classification. One of them was mentioned by Nordin, et al., and Diméglio, et al. [28,32]. They classified the lesion into four degrees as follows:

1. Degree 1, benign “postural or positional” clubfoot: The position of the foot is easily corrected through casting and physiotherapy.

2. Degree 2, moderate “soft more than stiff” clubfoot: This degree accounts about 33% of cases. It responds to casting in more than 50% of cases. The others not responding to this line of treatment within 7-8 months may need surgical interference.

3. Degree 3, severe “stiff more than soft” clubfoot: It occurs in 61% of cases. More than 50% of them don’t respond to conservative treatment and are mostly released surgically.

4. Degree 4, very severe “stiff” clubfoot: It is irreducible; and congenital anomaly. It often occurs in both sides; and necessitates extensive surgical repair (Table 1).

Grade Score Type Reducibility
I < 5 Benign > 90% soft-soft, resolving
II 5 to < 10 Moderate > 50% soft-stiff, reducible, partly resistant
III 10 to < 15 Severe < 50% stiff-soft, resistant, partly reducible
IV 15 to < 20 Very severe < 10% stiff- stiff, resistant

Table 1 Diméglio, et al., classification of congenital talipes equinovarus [32].

Cummings and Lovell mentioned another three degrees of severity [33]:

1- The first “mild” one is called postural clubfoot and needs no great effort in correction.

2- The second degree, called moderate clubfoot mostly does not need surgical interference. The foot is easily flexible with absence of the transverse crease. Cases of this degree usually respond to realignment followed by keeping the foot for a while in plaster cast.

3- The last “severe” degree is called defiant clubfoot. It is characterized by a small foot and tight skin with a transverse crease in the sole. The heel isn’t easily identified because of the fatty tissue covering the calcaneus. Fortunately, this type is less common than the previous type; but is resistant to conservative treatment and surgical interference is nearly inevitable.

Another common scoring or classification has been proposed by Pirani, et al. [34]. They divided the cases into three categories according to the six clinical findings; and gave three scores from 0-1 to each, as follows: 0 for absence, half for mild and one for presence of fixed contracture. The investigated signs are related to the midfoot and hindfoot. Those related to the midfoot are: 1- Curve of the lateral border of foot, 2- Presence of medial foot crease, & 3- Palpation of the head of talus laterally. The hindfoot signs are: 1- Posterior foot crease, 2- Palpation of heel, & 3- Equinus rigidity (Table 2). The clinical investigation is done through looking, feeling and moving the foot.

Foot

Clinical examination

Midfoot Hindfoot
Signs Score Signs Score
Looking Lateral border No deviation from straight line 0 Posterior crease No heel crease 0
Medial deviation distally 0.5 Mild heel crease 0.5
Severe deviation proximally 1 Deep heel crease 1
Feeling Head of talus Reduced talonavicular joint 0 Empty heel sign Hard heel (calcaneus is in normal position) 0
Subluxed but reducible talonavicular joint 0.5 Mild softness 0.5
Irreducible talonavicular joint 1 Very soft heel (calcaneus is not palpable) 1
Moving Medial crease No medial crease 0 Rigidity of equinus Normal dorsiflexion 0
Mild medial crease 0.5 Foot is plantigrade with knee extended 0.5
Deep crease altering contour of foot 1 Fixed equinus 1

Table 2 Pirani scoring of clubfo

The high Pirani score, the more severe clubfoot, i.e. score of six means severe case, while zero is of a normal foot [8].

Neglected clubfoot

Although clubfoot is a major concern for the parents and the whole family as it is a major obvious crippling defect affecting the walking of the child, many cases especially in developing countries are neglected. This neglect might be due to difficulty to gain medical care, shortage in skilled surgeons or ignorance of the parents. In these cases, the children are forced to walk on the dorsolateral side of the foot. The affected cases are unable to wear the shoes in their feet aggravating the problem. Neglecting cases are associated with sequelae and complications [35]. The affected neglected foot is usually reduced in size. This is caused by tethering of the abnormal tight ligaments and tendons that tightly catch the foot impeding its further growth. The clubfoot is often associated with a limitation or even absence of the movements in the subtalar and midtarsal joints leading to their stiffness [36]. The complications include pain, thickened pigmented skin and ulceration. Such ulceration could result in osteomyelitis that might lead to amputation of the foot at the end stages [37].

Management of Clubfoot

The goals of management of clubfoot are to obtain an obvious plantigrade, stable and straight foot with no need to modify shoes and a good range of foot motion without pain [5].

From the beginning of the 20th century till now, the management of clubfoot has fluctuated between conservative and surgical procedures. In the early 20th century, treatment was begun for older children; therefore, it was difficult and resistant to correct [38]. Treatment of clubfoot should start soon after birth; initiated within the first week of life. This is because the joints and bones of the infant’s foot are more flexible to allow successful repair. Treatment depends on the clubfoot degree. Mild flexible clubfoot is usually treated by non-operative or conservative treatment. Surgical treatment is reserved for cases not responded to the first line of treatment and for severe rigid clubfoot [39].

Conservative treatment

History of conservative treatment starts since Hippocrates in 400 BC. Conservative measures could be divided into two phases pre- and post-Ponseti eras. The importance of the Ponseti technique does not mean exclusion of other lines of treatment including surgery depending on the condition of each case [9].

There are many methods for conservative treatment; however, the International Clubfoot Study Group, established in 2003, has approved only three of them as the standardized conservative lines for the treatment of clubfoot; Kite, Ponseti and Bensahel techniques [9].

Kite’s technique

This method was introduced in the USA by Dr. Kite in the 1930s. It assumes gradual correction of each of the main deformities separately instead of simultaneous repair. The method depends on a series of manipulations and castings. To start in the next step, the previous one should be completely repaired. The technique starts with correction of med-tarsal adduction, internal rotation and calcaneal varus; and lastly the equinus. The session of manipulation is about five minutes, followed by lower limb immobilization [40]. The immobilization is performed through doing a slipper cast extending to a level below the knee that is changed every week. At the end of the procedure the foot is put in a Denis Browne Bar. The success of this method in various studies ranged from a low as19% to high up to 90% [9]. This method has not been performed longer because of long term treatment, with casting over two years as well the unsatisfactory outcomes in more than 50% of cases [41].

Ponseti method

The method was developed based on extensive anatomical study of the foot. It involves serial manipulations and casting, followed by a further three weeks in a cast. This method is widely adopted as the method of choice in many centers all over the world; however, most of the treated cases have a residual equinus and necessitate tenotomy of the calcaneal tendon. Tenotomy is usually indicated when the hindfoot can’t achieve dorsiflexion for 15 degrees after correction [42].

Relapses can be frequent after treatment by the Ponseti method. Such relapses need the child to wear an abduction brace for three months. Thereafter, the abduction brace is advised to be worn by the child only at night till reaching the age of four years to avoid the relapse. The deformity could be corrected by applying counter pressure on the head of the talus during application of the casts with keeping the foot in abduction and lateral rotation [6]. Failure rate of the Ponseti method accounts for 3-5% of cases; and this needs surgical interference [43].

The parents are recommended to be aware that the treatment by this method extends up to at least four years; and this needs their cooperation and serious commitment throughout the process of management.

Bensahel ‘French’ technique

This method was suggested in France in the 1970s; and entered English literature in the 1980s. It depends on daily sessions of physiotherapy manipulations of the infant’s clubfoot for thirty minutes for two months. This is followed by stimulation of muscles at the foot, especially the peronei, to keep the passive reduction achieved by physiotherapy manipulations; and then catching and holding the foot in the new position by adhesive strips. This daily management is reduced to three sessions per week till the age of six months. Then, taping of the foot continues until the infant begins walking. Thereafter, the foot is supported with a splint at night for another two to three years. About half of cases are totally improved; and the other cases only require simple surgical interference through a posterior release. Disadvantages of this method include the long-term management for many years that necessitates close contact of the child and his parents with the hospital [9]

Richards, et al.. compared the outcome results of non-operative techniques of the Ponseti and French methods [44]. They concluded that non-significant better results were achieved by the Ponseti method; and added that the poor results were the same in both methods accounting for about 16% of cases in each one. However, He, et al., analyzed the clinical outcome of the different conservative measures of clubfoot treatment in a total 1435 cases [45]. They recommended the Ponseti method to be the first method of choice for management of such cases. They added that this method gives better results than the other conservative techniques; and minimizes numbers of the cases requiring surgical intervention.

Complications of conservative treatment

The main complications are summarized in two points; the first is the recurrence or failure of treatment while the second is the false correction leading to a condition called ‘rocker-bottom foot’. In such a situation, there is overstretching of the foot without actual correction of the equinus of the hindfoot, but the foot tends to take up position towards the neutral position. If this is detected during treatment, manipulations should be stopped in particular to equinus correction; and the foot is rested in a supporting splint in equinus to allow healing of breach in the midfoot. Otherwise, more disability and pain will occur with more difficulty treating the deformed foot [14].

Surgical treatment

The best time for surgical interference for treating the clubfoot is still controversial. Many authors suggest that clubfoot is best to be operated at 9-10 months. At this age, the infant begins to pull himself to attain standing and hence beginning to put body weight on the feet. This might benefit from the gravity in the process of repair [14]. Despite other authors agreeing to perform surgery as early as possible, no evidence of better results to those operated early in life has been found [46]. The authors added that in very young ages, there may be difficulty to recognize the small bones and other cartilaginous structures in addition to presence of abundance of fatty tissues. Surgery in such a condition requires a meticulous surgeon to avoid residual scarring and stiffness resulting from dealing with immature structures. Therefore, surgeries must be performed by expert surgeons at specialized clubfoot centers established for such purposes especially in large hospitals receiving thousands of births per year [14].

The list of types of operations performed to correct clubfoot that begin at 1891 till now is endless; and no single one gives long-lasting repair [9].

The surgery in the current use can be divided into three categories; the first involving soft tissue, the second with bones and the last including both soft tissue and bones. Operations including bones are usually done for children at an older age; and are considered salvage procedures [6].

I. Soft tissue surgery

This procedure includes release, lengthening or transfer of tight structures such as ligaments and tendons and/or deforming soft tissue structures, e.g. joint capsules. The deformity should be corrected before further surgery [28].

Posterior release is the simplest soft tissue release surgery. It includes lengthening of the tendoAchilles and capsulotomy of the talocrural and subtalar joints as well as cutting the posterior talofibular and calcaneofibular ligaments. Such ligaments act as a tether for the talus and calcaneus so their contracture might prevent normal foot dorsiflexion [46].

Other comprehensive soft tissue release could involve posteromedial release of the soft contractures of the posterior, medial and subtalar soft tissues. This might allow correct alignment of bones. Also, circumferential release and posterior or tibialis tendon transfer might be performed to permit dynamic balance between the invertor and evertor muscles [28,33].

II. Combined skeletal and soft-tissue procedures

Evan proposed a procedure depending on shortening the lateral column of the foot in order to realign the midtarsal joint [47]. The author performed it through a closing-wedge resection of the calcaneocuboid joint associated with a modified soft tissue release of medial aspect of the foot.

Lunderg mentioned performing an opening-wedge osteotomy through the calcaneus in addition to the insertion of a bone wedge [48]. This is concomitant with posteromedial soft tissue release in order to obtain full repair.

Ilizarov found that gradual distraction of structures e.g. soft tissues and bones especially in young ages could lead to cellular proliferation of such structures [49]. The author stated that using the Ilizarov external fixator for management of clubfoot affords correction at many planes. Other authors investigated such methods with soft tissue release and bone procedures; and concluded that this method might give good results in management of neglected and relapsed clubfoot [50].

III. Skeletal surgery

There is a general consensus that surgery on the bones of the foot is reserved for older children or cases resistant to other measures of treatment [5].

Management of neglected and severe resistant rigid clubfoot represents a great challenge to orthopedic surgeons. This is because the excessive and extensive open surgical manipulations could lead to postoperative scarring and many complications. Therefore, talectomy has been suggested to be a salvage surgical method for correction of such cases of clubfoot [3].

Talectomy

Currently, there is an increasing attention all over the world to the role of talectomy in correction of cases of rigid clubfoot not responding to other measures [3].

Talectomy has been suggested as a salvage procedure to manage severe resistant cases of clubfoot. The operation gives satisfactory results through removing the talus “the most distorted bone”. The patient could wear shoes with a plantigrade foot following talectomy. It is a safe and one-step surgery without major complications [30].

Conclusions

Clubfoot is a challenging orthopedic problem especially in severe resistant cases. Such cases are mostly encountered associated with other congenital anomalies. Thorough investigation of cases to exclude the association of other anomalies is essential to determine the line of treatment. Therefore, it is suggested to perform talectomy as a salvage procedure in cases of failure of other conservative measures; and it is also suggested as the first line in cases of severe resistant clubfoot associated with other congenital anomalies. Future studies are also recommended particularly to reveal genetic involvement in its etiology. This might be of benefit to alleviate or minimize occurrence of clubfoot.

Conflict of Interest: None

Funding: None

Acknowledgments

We would like to thank ALL members of the Orthopedic Surgery Department, Zagazig University. All diagrams are made by the corresponding author “A A Hegazy”.

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  29. Graf A, Wu K, Smith PA, Kuo KN, Krzak J, Harris G: Comprehensive review of the functional outcome evaluation of clubfoot treatment: a preferred methodology. J Pediatr Orthop B. 2011;21:20-27. 10.1097/BPB.0b013e32834dd239
  30. Hegazy MA, Khairy HM, El-Aidy SM: Role of Talectomy in Severe Resistant Clubfoot in Children. J Foot Ankle Surg (Asia Pacific) 2019;6:29-38. 10.5005/jp-journals-10040-1105
  31. Miyagi N, Lisaka H, Yasuda K, Kaneda K: Onset of ossification of the tarsal bones in congenital clubfoot. J Pediatr Orthop. 1997;17:36-40.
  32. Diméglio A, Bensahel H, Souchet P, Mazeau P, Bonnet F: Classification of clubfoot. J Pediatr Orthop B. 1995;4:129-136. 10.1097/01202412-199504020-00002
  33. Cummings RJ, Lovell AW: Current concept review: Operative treatment of congenital idiopathic club foot. J Bone Joint Surg. 1988;70:1108-1112.
  34. Pirani S, Hodges D, Sekeramyi F. : A reliable and valid method of assessing the amount of deformity in the congenital clubfoot deformity (The Canadian Orthopaedic Research Society and the Canadian Orthopaedic Association conference proceeding) . 2008;90-B(Suppl I):53.
  35. Penny JN: The neglected clubfoot. Techniques Orthop. 2005;20:153-166. 10.1097/01.bto.0000162987.08300.5e
  36. Sobel E, Giorgini R, Velez Z: Surgical correction of adult neglected clubfoot: Three case histories. J Foot Ankle Surg. 1996;35:27-38. https://doi.org/10.1016/S1067-2516(96)80009-3
  37. Mirzayan R, Early SD, Matthys GA, Thordarson DB: Single-stage talectomy and tibiocalcaneal arthrodesis as a salvage of severe, rigid equinovarus deformity. Foot Ankle Int. 2001;22:209-213. 10.1177/107110070102200307.
  38. Ippolito E, Farsetti P, Valentini MB: Management of Clubfoot. In: Bentley G. (eds) European Surgical Orthopaedics and Traumatology. Springer, Berlin, Heidelberg; 2014. https://doi.org/10.1007/978-3-642-34746-7_157
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  44. Richards BS, Faulks S, Rathjen KE, Karol LA, Johnston CE, Jones SA: A comparison of two nonoperative methods of idiopathic clubfoot correction: the Ponseti method and the French functional (physiotherapy) method. J Bone Joint Surg Am. 2008;90:2313-2321. 10.2106/JBJS.G.01621
  45. He JP, Shao JF, Hao Y: Comparison of different conservative treatments for idiopathic clubfoot: Ponseti’s versus non-Ponseti’s methods. J Int Med Res. 2017;45:1190-1199. 10.1177/0300060517706801
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Congenital amniotic band constriction of the proximal tibia: A Yucatan project case report

by Alexandra Heidtmann, BS1; Lahari Madulapally, BS, MA1; Luis Rodriguez Anaya, DPM2*; Daniel Cawley, DC, MS2

The Foot and Ankle Online Journal 13 (4): 9

The Yucatan peninsula, in southeastern Mexico, is home for a high incidence of various physical and neurological deformities. This provided Dr. Charles Southerland of Barry University School of Podiatric Medicine to establish the Yucatan Crippled Children’s Project with the main goal of providing care for a community with limited resources. Over the years, the project has evolved into a crucial tool of academics and medical research for students and physicians and is partially sponsored by the International Foot & Ankle Foundation for Education and Research [1]. This clinical case report from the Yucatan Project addresses a rare condition of an in utero fibrous band composed of amniotic fluid constricting the proximal tibia of the fetus.

Keywords: amniotic band, proximal tibia, Z-plasty

ISSN 1941-6806
doi: 10.3827/faoj.2020.1304.0009

1 – Fourth Year Medical Student at Barry University School of Podiatric Medicine, Miami, Florida.
2 – Assistant Professor Barry University School of Podiatric Medicine
* – Corresponding author: LARodriguez@barry.edu


Proper development of the embryo involves a delicate sequence of spatiotemporal gene expression that governs tissue development and pattern formation [2]. At the beginning of the 2nd week of development, the blastocyst begins the process of implantation within the uterine wall. The embryoblast reorganizes into a bilaminar disc made up of two cell layers, the epiblast, and hypoblast [3]. The embryo becomes covered by two thin membranes derived from extraembryonic ectoderm and mesoderm, the amnion and the chorion. Fluid accumulates between the epiblast and the trophoblast cells lining the wall of the blastocyst, creating the amniotic cavity. Epiblast lines the new cavity forming the amniotic membrane. The extraembryonic mesoderm, derived from the hypoblast and primary yolk sac, proliferates and fills the cavity of the blastocyst. The extraembryonic mesoderm surrounding the yolk sac splits into two layers, creating the chorionic cavity [4]. The chorionic cavity separates the embryo and amniotic cavity from the outer wall of the blastocyst which is lined by the chorionic membrane. (Larsen, 2015). The amnion produces fluid causing expansion of the amniotic cavity, eventually obliterating the chorionic cavity. This results in the fusion of the amnion with the chorion [5]. Folding of the embryonic disc pulls the amniotic cavity ventrally, enclosing the developing embryo within the amniotic sac (fused amnion and chorion). This allows the embryo to be suspended in a liquid environment which will allow for growth while protecting against mechanical injuries and adhesions [3, 6].

Development of the lower limb involves limb positioning and outgrowth, rotation, and the development of region-specific morphology. These events are governed by gene expression and morphogen gradients [3, 7]. The lower limb buds begin to appear late in the 4th week of gestation on the ventrolateral body wall, opposite the lumbar somites. The lower limb buds consist of an undifferentiated mesenchymal (mesoderm) core, derived from the lateral plate mesoderm, covered by an epithelial layer derived from ectoderm. The epithelium over the distal margin of the limb bud thickens forming the Apical Ectodermal Ridge (AER). The AER is a major signaling center that guides the proliferation and elongation of the underlying mesoderm through reciprocal signaling [7].

The limb bud core initially contains cells from the lateral plate mesoderm that will give rise to the skeleton, vasculature and the connective tissue of the future limb. Migrating neural crest cells will give rise to the sensory nerves, Schwann cells, and melanocytes [3]. During the 4th week, myogenic mesodermal cells begin migrating from the somites into the lower limb bud, creating the flexor and extensor muscle masses that will surround the future bone. As the limb bud enlarges, muscle mass will increase through mitosis until the mid-fetal period. During the 5th week, the paddle-shaped foot plates develop. Skeletogenesis begins during the 5th week establishing models for future limb bones. During the 6th week, lower limb segments become recognizable and most of the mesenchymal models begin the process of chondrification. By week 7, the digital rays of the foot plate become visible. Primary ossification of the cartilaginous templates begins as early as the 8th week in the lower limb [8, 9]. Vascular supply to the embryonic limb bud is primarily through the axial artery. During the 8th week, the axial artery involutes and the femoral artery system becomes the major vascular supply to the lower extremity, establishing the adult pattern of vasculature. Between the 6th to 8th week, the limb buds undergo long axis medial rotation and assume a more ventral position on the body wall. By week 7, the feet are located primarily in the sagittal plane. The tibial (pre-axial) borders are oriented cephalically with the extensor/dorsal surfaces facing laterally and the flexor/plantar surfaces facing medially. Rotation continues through the fetal period, eventually orienting the knees cephalically the extensor surface anteriorly/superiorly and the flexor surface posteriorly/inferiorly. During the fetal period, the thigh continues to rotate internally, while the foot dorsiflexes and pronates [4, 10].

Background on Congenital Amniotic Bands

Congenital amniotic band constriction, also known as amniotic band syndrome and Streeter dysplasia, was first defined by Montgomery in 1832 [11]. The two proposed explanations are the intrinsic and extrinsic theories. The intrinsic theory, proposed by Streeter, describes the composition of the bands as tissue that has been left behind due to a defect in the development of germ plasm [11]. However, the most widely accepted pathophysiology is the extrinsic theory proposed by Torpin in 1965 [12]. This theory explains that the fibrous bands occur due to the amnion separating from the chorion. The amnion further wraps around a part of the embryo constricting its growth and causing malformations. As previously mentioned, the amnion is responsible for creating amniotic fluid. Therefore, the developmental injury leads to a deficiency in amniotic fluid that contributes to the displacement of fetal limbs into the chorionic cavity, where compression occurs [12].

Patterson’s classification divided the stages of these congenital amniotic bands based on the severity and outcomes of the constriction [13]. Stage A is classified as simple constriction rings. Stage B describes constriction rings with deformities of distal aspects of limbs with or without lymphedema. Stage C involves constriction rings with fusion of distal limbs, such as acrosyndactyly. Lastly, Stage D is the most severe and leads to intrauterine amputation. Additionally, two or more of these symptoms are needed to diagnose congenital amniotic band constriction. This classification system can provide insight into proper treatment options according to the extent of the constriction [13].

Although no concrete etiology has been identified, literature described a few possible causes of congenital amniotic bands such as local disruption of vasculature, constricted uterine environment caused by oligohydramnios or large benign tumors of the uterine myometrium.

Constriction bands can lead to a myriad of problems such as vascular compromise leading to ischemia and venous congestion. More severe cases have been reported to cause natural limb amputation, pseudosyndactylism, anencephaly, craniofacial abnormalities and abdominal wall defects [14, 15].

Clinical Presentation

A 4-year-old female presented to the Yucatan Project clinic in February of 2014. Neonatal records were not provided. Upon physical examination, a congenital band constriction was noted to the proximal tibia of the right leg. Skin texture is smooth with no signs of infection, hyperkeratosis or xerosis. Non-pitting edema was present in the right knee and leg both proximal and distal to the constriction band as seen in Figure 1a and 1b.

Figure 1 a: Weight bearing anterior clinical view, showing right leg constriction band, and b: Non weight bearing clinical view.

Figure 2 a: 60 degree Z-Plasty and, b: Multiple Parallel Z-Plasty.

According to the Patterson classification system, this patient would be categorized into Stage B with lymphedema.

Surgical Procedure

Continuous parallel multiple stereometric Z-plasty surgical procedures were performed March of 2014 with the purpose of releasing the constriction band. The first step of the Z-plasty is to draw a line for the central arm incision directly along or parallel to the skin contracture. The additional two arms of the Z-plasty are then added to the edges of the central arm, forming a 60-degree angle between the central and additional arms. With a 60-degree Z-plasty, 75% lengthening should be achieved, as seen in Figure 2a. The length of the central arm should also have the same length as the two additional arms.

The skin dissection should only go with the extent of the deep fascia, preserving the vascular supply of the two newly formed triangular flaps, as shown in Figures 3 and 4 [16, 17]. The Z incision then allows for transposition of the triangular flaps to gain length along the longitudinal axis at the expense of the transverse skin narrowing.

Figure 3 Intraoperative image following skin incision.

Figure 4 Intraoperative image following Z-Plasty.

The multiple parallel Z-plasty are connected throughout the contracture line as seen in Figure 2b [18].

In November of 2014, the patient presented to the clinic for a 9-month postoperative follow-up visit. The incision site was healing well with no signs of infection. Edema was significantly reduced on the right proximal tibia where the surgery was performed. Mild discoloration was present at the scar site, as seen in Figure 5.

Figure 5 Nine month postoperative view of the right leg.

The patient was noted to have mild residual foot drop on the right side, most likely due to damage of the common peroneal nerve from the previous constriction band or the surgical procedure itself.

Discussion and Results

Congenital amniotic band constriction can occur in any area of the body due to disruptions in fetal development. This deficiency in amniotic fluid causing the displacement of body parts can be limb or life-threatening. If detected early, proper care can be given to avoid drastic results.

In the literature, several surgical procedures have been reported to help patients that suffer from congenital amniotic band constriction: Z-plasty, W-plasty, syndactyly release, skin and nerve grafting, and tendon transfers.The Z-plasty, as described above, still remains as the main skin lengthening surgical correction. The W-plasty is a description of multiple W-plasties, which may be necessary when the constriction is severe in order to effectively reduce the lymphedema [19]. Syndactyly or acrosyndactyly has been reported as a common complication of these bands, in which a syndactyly release has been performed. Skin grafting has been utilized to cover the area of tissue loss caused by syndactyly release [11]. Following the aforementioned surgical procedures, one of the postoperative complications is nerve paralysis. A segment of the sural nerve can be used to replace the damaged nerve. Lastly, tendon transfers have helped to recover muscle function of previously limited range of motion (9] .

Numerous lower extremity issues can also arise due to these congenital amniotic bands. In a study that involved 83 patients with congenital amniotic band constriction, 19 patients developed clubfoot deformities. Of these 19 patients, 10 patients had compression of the common peroneal nerve which led to paralysis of the clubfoot deformity [20]. Another case report identified a patient that had amniotic band constriction of both left and right lower extremities. The left lower extremity was constricted at the distal tibia and fibula to the point of autoamputation and there was no development of bones in the foot. A below the knee amputation had to be performed to the left lower extremity, as it was deemed unsalvageable. The right lower extremity constriction led to lateral bowing of the tibia and fibula, as well as a clubfoot deformity. A W-plasty and Z-plasty was performed on the right lower extremity to release the underlying tissue. The patient was eventually able to ambulate with the help of a prosthesis on the left side [21].

In the case report presented in this paper, the constriction band at the proximal tibia was categorized as Stage 2 according to Patterson’s classifications. It did not lead to any malformations of the distal aspects of the lower extremity. The patient received a Z-plasty procedure and was able to ambulate with a mild residual foot drop. A future surgical option for the patient can be a sural nerve graft to replace the damaged common peroneal nerve as an attempt to recover dorsiflexion at the right ankle joint. Due to the lack of documentation provided with this patient, it is difficult to formulate the reasons that may have caused these amniotic constriction bands. Additionally, the patient did not return for further follow-up visits, which prevented documentation of the patient’s progress or other complications of the surgery.

Conclusions

Congenital amniotic constriction band is an in utero fibrous band that constricts the fetus potentially resulting in limb and life-threatening conditions. Identifying the presence of constriction bands early in pregnancy can reduce dangerous effects. However, if the condition is not detected during pregnancy, surgical intervention is necessary to prevent further complications for the patient. Due to the rarity of congenital amniotic bands, documentation of affected patients is crucial to further expand health care professional’s knowledge on the condition and treatment options.

References

  1. Internationalfootankle.org [internet]. International Foot and Ankle Foundation for Education and Research; 2020 [cited 2020 Sep 1]. (www.internationalfootankle.org).
  2. Assou S, Boumela I, Haouzi D, et al. Dynamic changes in gene expression during human early embryo development: From fundamental aspects to clinical applications. Hum Reprod Update. 2010 Aug 17;17(2):272-90.
  3. Carlson BM. Human embryology and developmental biology. 5th ed. Philadelphia: Saunders; 2013 Mar 6.
  4. Schoenwolf GC, Bleyl SB, Brauer PR, Francis-West PH. Larsen’s Human Embryology. Churchill Livingstone; 2014 Nov 25.
  5. Filly RA. Ultrasound evaluation during the first trimester. In: Callen PW. Ultrasonography in Obstetrics and Gynecology. 3rd ed. Philadelphia: Saunders, 1994;63.
  6. Hill, MA. Embryology, amniotic cavity development. 2019.
  7. Barham G, Clarke NMP. Genetic regulation of embryological limb development with relation to congenital limb deformity in humans. J Child Orthop. 2008 Feb 7;2(1):1-9.
  8. Ogden JA. Development of the lower limb. Congenital Lower Limb Deficiencies. New York: Springer; 1989.
  9. Harkness LM, Baird DT. Morphological and molecular characteristics of living human fetuses between Carnegie stages 7 and 23: developmental stages in the post-implantation embryo. Hum Reprod Update. 1997; 3(1):3-23.
  10. Kelikian AS, Sarrafian SK. Sarrafian’s Anatomy of the Foot and Ankle: Descriptive, Topographic, Functional. 3rd ed. Philadelphia: Wolter Kluwer Health/Lippincott Williams & Wilkins. 2011; p. 144-154.
  11. Adu E, Annan C. Congenital constriction ring syndrome of the limbs: A prospective study of 16 cases. Afr J Paediatr Surg. 2008;5(2):79-83.
  12. Cignini P, Giorlandino C, Padula, F, Dugo, N, Cafà EV, Spata A, et al. Epidemiology and risk factors of amniotic band syndrome, or ADAM sequence. J Prenat Med. 2012;6(4):59–63.
  13. Patterson T. Congenital ring-constrictions. Br J Plast Surg. 1961 Apr;14, 1–31.
  14. Rezai, Shadi, Faye, Justin, Chadee, Annika, et al. Amniotic Band Syndrome, Perinatal Hospice, and Palliative Care versus Active Management. Case Reports in Obstetrics and Gynecology. 2016.
  15. Sentilhes L, Verspyck E, Eurin D, Ickowicz V, Patrier S, Lechevallier J, Marpeau L, et al. Favourable outcome of a tight constriction band secondary to amniotic band syndrome. Prenat Diagn. 2004 Feb 27; 24(3).
  16. Southerland JT. McGlamrys Comprehensive Textbook of Foot and Ankle Surgery. 4th ed. New York: Lippincott, Williams & Wilkins. 2012.
  17. Chicko B, Pollard S, Lee H. Crozer-Keystone Residency Manual. Crozer Keystone Health System. 2011.
  18. Dockery GD. Z and W skin plasty. In, Dockery G, Crawford ME (eds), Lower extremity soft tissue and cutaneous plastic surgery. 2nd Ed. Philadelphia: Lippincott Williams & Wilkins; 2012. p. 163-176. https://www.sciencedirect.com/book/9780702031366/lower-extremity-soft-tissue-and-cutaneous-plastic-surgery
  19. Light TR, Ogden JA. Congenital Constriction Band Syndrome Pathophysiology and Treatment. Yale J Biol Med. 1993 May-Jun; 66(3), 143–155.
  20. Tada K, Yonenobu K, Swanson AB. Congenital Constriction Band Syndrome. Journal of Pediatric Orthopaedics, 1984 Oct 21;4(6),726–730.
  21. Magee T, Mackay DR, Segal LS. Congenital constriction band with pseudoarthrosis of the tibia: a case report and literature review. Acta Orthop Belg. 2007;73(2), 275-278.

 

 

 

Distal lower extremity manifestations in spina bifida patients of the Yucatan Peninsula: A 24-year retrospective case series

by Alexandra Heidtmann, BS1; Lahari Madulapally, BS, MA1; Luis Rodriguez Anaya, DPM2*; Daniel Cawley, DC, MS2

The Foot and Ankle Online Journal 13 (4): 8

Spina Bifida, a rare congenital disorder with an incidence of 7.85 per 10,000 births in Mexico. It results from the failed closure of the neural tube leading to the incomplete development of the neural arches. This case series is part of the Yucatan Crippled Children’s Project that began in 1996 by Charles Southerland, Doctor of Podiatric Medicine and former professor of Barry University’s School of Podiatric Medicine. All patients in this study were assessed and treated at the Red Cross hospital in the city of Merida, Yucatan, Mexico. Attendings, residents and medical students travel to the Yucatan Peninsula four times a year for a period of one week. Given that this study was built from reports of medical mission trips that occur four times a year with limited resources and time, the lack of documentation of treatment plans and follow-ups made it difficult to identify surgical procedures and assess the success of surgeries. Additionally, we did not have access to the patients birth records or their mothers medical records to accurately determine the etiology of their deformities. Based on our data, we conclude that intervention should be considered as early as possible in any flexible deformity to prevent them from becoming rigid.

Keywords: Spina Bifida, talipes equinovarus, clubfoot, adductovarus, calcaneo valgus, Ponseti, osteomyelitis

ISSN 1941-6806
doi: 10.3827/faoj.2020.1304.0008

1 – Fourth Year Medical Student at Barry University School of Podiatric Medicine, Miami, Florida.
2 – Assistant Professor Barry University School of Podiatric Medicine
* – Corresponding author: LARodriguez@barry.edu


Spina Bifida, a rare congenital disorder with an incidence of 7.85 per 10,000 births in Mexico. It results from the failed closure of the neural tube leading to the incomplete development of the neural arches [1]. Spina bifida is the result of genetic and non-genetic factors that interfere with the folding and closure of the neural tube. In its most severe form, meningomyelocele, the neurons of the spinal cord are exposed to amniotic fluid resulting in neuronal death. In addition, the spinal cord and meninges protrude through the midline bony defect of the back [2].

The clinical manifestation of a meningomyelocele is dependent on the spinal level of involvement and the presence of cerebral involvement and hydrocephalus [3]. Sensory and motor impairments are commonly present below the level of the lesion causing alterations in the bowel and bladder function, muscle paresis and paralysis, and sensory loss. Impairment is classified by the level of neurosegmental involvement determined by the strength of specific muscle groups [3]. Nearly all patients with spina bifida will experience manifestations in their feet, especially those cases involving the thoracic and lumbar spinal regions [4,5]. Previous studies have reported that the most common manifestation of spina bifida in the feet are talipes equinovarus, equinus, vertical talus, calcaneal deformities, and cavovarus [4,6,7]. The aim of this study is to analyze the incidence of various distal lower extremity manifestations and their long-term effects on spina bifida patients of the Yucatan Peninsula.

Methods

This case series is part of the Yucatan Crippled Children’s Project that began in 1996 by Charles Southerland, Doctor of Podiatric Medicine and former professor of Barry University’s School of Podiatric Medicine [8]. All patients in this study were assessed and treated at the Red Cross hospital in the city of Merida, Mexico. Attendings, residents and medical students travel to the Yucatan Peninsula four times a year for a period of one week.

From 1999 to 2020, we retrospectively analyzed 1,489 patients that were seen by physicians from the Yucatan Crippled Children’s Project. Among the total, we identified 25 patients, 17 male and 8 female, with history of Spina Bifida and concomitant lower extremity deformities. From the 25 patients, 15 patients had bilateral lower extremity deformities and 10 patients had unilateral deformities, leading to a total of 40 limbs. The ages ranged from 3 months old to 43 years old, with a total average age of 11.33 years. The mean age for rigid deformities was 15.4 years, while the mean age for flexible deformities was 8.8 years.

We analyzed 3 cases of patients with a history of spina bifida and lower extremity deformities according to the clinical notes collected from the Yucatan Crippled Children’s Project.

Case Presentation 1

Case 1 is a 2-year-old male who presented to the Yucatan Crippled Children’s project clinic in March of 2007 with a chief complaint of difficulty ambulating. Patient’s family reports past medical history of birth at 38 weeks and significant time spent in the NICU due to hydrocephalus and spina bifida. Upon initial assessment, the patient was alert and oriented and showed no additional symptoms. Patient was diagnosed with bilateral flexible clubfoot deformity, as seen in Figures 1 and 2. Considering the age and the flexibility of the deformity, the conservative Ponseti serial casting technique was performed on the patient.

Figures 1 and 2 Plantar and dorsal views of bilateral clubfoot prior to Ponseti method.

Figures 3 and 4 Patient at 12 years old, 10 years after Ponseti method. AP Radiograph of bilateral feet. Pre-operative clinical picture of bilateral feet.

Figures 5, 6, 7 Intraoperative picture of left foot before and after external fixator application. AP radiograph of left foot.

The patient and his family were instructed to follow – up with the local doctor. The patient returned to the clinic in November of 2017, at 12 years old, with chief complaint of continued difficulty ambulating due to the progression of the windswept deformity (Figures 3 and 4). After assessment, the left foot was diagnosed with adductovarus and talipes equinovarus deformity. The right foot was diagnosed with forefoot adduction, midfoot abduction, and calcaneovalgus which are the three components of complex skew foot. Given the Ponseti technique applied ten years ago had failed, the deformity has worsened and progressed from flexible to rigid. The procedure consisted of application of external fixation with medial motor for gradual correction of adductovarus deformity on the left foot (Figures 5-7).

Figure 8 Bilateral flexible cavovarus deformity with ulcer on dorsolateral aspect of right foot.

Figures 9 and 10 Nine year follow-up shows rigid bilateral cavovarus deformity. The patient is confined to a wheelchair.

The patient presents to the clinic in February of 2018 for a 3 month postoperative visit after application of external fixation. It was noted that the toes were not fixated during the external fixator surgery and they developed flexion contractures within the reduction frame. The patient developed clinodactyly of all five digits of the left foot. At this date, the frame was removed and the patient began physical therapy in an attempt to reduce flexion contractures.

Case Presentation 2

Case 2 is an 8-year-old female with a past medical history of spina bifida and sensory neuropathy bilaterally. The patient presented to the clinic in July of 2011 with a chief complaint of a wound on the right foot. Upon physical exam, an open ulcer was noted on the dorsolateral aspect of the right foot along the cuboid-5th metatarsal joint (Figure 8). The wound has a beefy red base with friable granulation tissue and a circumferential macerated periwound with suspected areas of hyperkeratotic tissue. Mild hyperpigmentation and erythema is noted proximally towards the dorsum of the ankle. The patient was diagnosed with a pressure ulcer on the right foot and bilateral flexible cavovarus deformity. Ulcer was managed during the patient’s first visit prior to any surgical intervention. Upon healing of the ulcer, surgery was performed; however, the surgical technique was not recorded.

The patient was virtually contacted during Covid-19 2020 Pandemic, and sent Figures 9 and 10. The patient stated she still has insensate feet and is unable to ambulate. Deformity has progressed to rigid and she is waiting until the next Yucatan Medical Mission Trip to possibly undergo another surgery that would allow her to ambulate.

Case Presentation 3

Case 3 is a 24-year-old male with a past medical history of spina bifida, insensate feet, and chronic lymphangitis. The patient presented to the Yucatan Project Clinic in April of 2005 with a chief complaint of wounds on the right foot. Upon physical exam, ulcer on the lateral aspect of the head of the 5th metatarsal of the right foot was noted to have a 50/50 granular fibrotic base with slough in the center. The periwound consisted of hyperkeratotic tissue on the plantar aspect of the 5th metatarsophalangeal joint. Hyperpigmentation is present extending proximally on the dorsum of the foot. The second wound, located on the lateral aspect of the 5th metatarsal tuberosity of the right foot, appeared to have 75/25 fibrotic granular base with regular borders. The patient was diagnosed with active infected ulcers and bilateral cavovarus deformity (Figures 11 and 12). Initial treatment consisted of ulcer debridement and offloading of the right foot with a 3D Walker. The patient was seen in November of 2005, 7 months after initial treatment. The right foot still remained dysfunctional with chronic non-healing wounds. Radiographs show radiolucency from mid-shaft of 4th and 5th metatarsal distally to the 4th and 5th distal phalanges and thickened periosteum of the proximal end of the mid shafts of 4th and 5th metatarsals on Figure 13, suggesting osteomyelitis.

Figures 11 and 12 Dorsolateral view of right foot showing ulcer along 5th metatarsal and medial view of the right foot showing cavovarus deformity.

Figure 13 AP Radiograph of bilateral feet.

Figures 14 and 15 Preoperative view of the right foot. Intraoperative picture of the right foot after Lisfranc amputation.

Figure 16 Dorsolateral view of right foot after amputation.

Due to the lack of access to other diagnostic tools, combined with the request from the patient for a permanent solution, a LisFranc amputation was performed on the right foot and osteoset beads with vancomycin were inserted to treat the infection (Figures 14 and 15).

The patient was seen in February of 2006, 3 months after LisFranc amputation of the right foot. The surgical site healed well with good results (Figure 16). However, the cavovarus deformity remained on the left foot (Figure 17).

The patient was seen again in November of 2006 for the last time, 12 months after LisFranc amputation of the right foot. The patient redeveloped an equinus deformity of the right foot and ulcers under styloid processes bilaterally (Figure 18). The patient was treated with well-padded plastazote ankle foot orthosis (AFO).

Figure 17 Dorsolateral view of left foot.

Figure 18 Ulcer under styloid process of the right foot after amputation.

Results

The most common lower extremity manifestation was ulcerations. In 17 ulcerated limbs, 8 were insensate and 5 developed osteomyelitis. Out of the 40 limbs, 5 ulcerated limbs had no reported gross deformity, and therefore were not included in the graphs. The remaining 35 limbs were biomechanically classified as rigid and flexible. In the 13 rigid limbs, there were 4 equinus, 3 talipes calcaneus, 7 cavus, and 2 planus feet. These were further subclassified into 6 more categories: equinovarus, pes cavocalcaneus, cavovalgus, cavovarus, pes planovalgus and no additional deformity. Rigid deformities subgroups can be seen in Graph 1. In the 22 flexible limbs, there were 9 equinus, 5 talipes calcaneus, 6 cavus, and 2 planus feet.

Graph 1 Rigid deformity of the foot.

Graph 2 Flexible deformity of the foot.

They were then subclassified into 5 more categories: calcaneovalgus, calcaneovarus, equinovarus, cavovarus, and no additional deformity. Flexible deformities are illustrated in Graph 2.

Discussion

Lower extremity manifestations due to spina bifida are difficult to be classified and the rate of misdiagnosis and mistreatment is high [9]. Similarly to previous findings, the most frequent foot deformity in our study was flexible equinus [4,5,10]. However, we found that rigid pes cavus was the second most predominant foot deformity in the Yucatan Peninsula, contrary to previous reports. Based on the limited medical access in the area and the higher average age of patients presenting with rigid deformities (15.4 years) when compared to flexible deformities (8.8 years), we suggest that this is possibly due to years of leaving the deformity untreated.

The prevalence of spina bifida was 7.85 per 10,000 births or 0.0785% in the country of Mexico [1]. However, in the Yucatan Peninsula the prevalence was found to be significantly higher. In this study, out of the 1,489 total cases analyzed from years 1999 to 2020, 25 patients with spina bifida were identified. This shows a prevalence rate of 167.90 per 10,000 births or 1.68%. Folic acid is a nutrient that is essential to the development of the fetus. Spina bifida and other birth defects form within the initial 28 days after conception. These congenital deformities can be prevented by ensuring sufficient blood folate levels in the mother during fertile years and early fetal development [11]. In the literature, North America has been shown to have the lowest incidence of spina bifida while Asia has the highest incidence. This could be due to Canada and the United States being the first countries to mandate folic acid fortification. In addition, even though mandatory fortification with folate has been implemented in many countries, it might not be enough folic acid to reach the daily recommended dosage of 400 micrograms. Therefore, it is important for mothers before conception and in the early fetal developmental months to supplement their folic acid intake [12].

Case 1 illustrated a patient with talipes equinovarus, also known as clubfoot deformity on the left foot. This triplanar deformity includes 3 components: ankle equinus, hindfoot varus, and forefoot adduction. Traditionally, there is a higher prevalence of clubfoot in males with a ratio of 2:1 to female and approximately 50% of the cases are bilateral. A few etiologies have been described in the literature, mainly divided into idiopathic and non-idiopathic. Idiopathic consists of limited intrauterine position due to a larger size of the fetus or smaller frame of mothers, while non-idiopathic includes a history of congenital deformities such as spina bifida, cerebral palsy, and meningitis. Clinical presentation of patient 1 at birth predisposed him to a higher risk of developing clubfoot given he is a male with a history of spina bifida. We do not have additional history of patient 1 such as birth weight and height, however, these factors could have also played a role in the patient developing clubfoot [13]. The Ponseti technique, a conservative treatment, was attempted when the patient was 2 years old; however, this technique is only proven to be successful in patients with flexible clubfoot up to 120 days of age [14]. On the right foot, the patient has had a long-standing complex skewfoot with forefoot adduction, midfoot abduction, and calcaneovalgus. The unsuccessful result of the Ponseti method on the left foot, combined with years of the patient not returning for medical assistance, led the bilateral deformity to become rigid on both feet.

Case 2 presented a 2-year-old female with history of spina bifida, insensate feet, active ulcer on the right foot, and flexible bilateral cavovarus deformity. Cavovarus involves a high longitudinal plantar arch, hindfoot varus, forefoot equinus, and pronated first ray in the stance phase of gait. If this deformity is present bilaterally, the most likely etiology is a neurological condition; however, if it is present unilaterally the etiology can be related to trauma such as pilon fractures or talar neck fractures [15]. In a flexible cavovarus foot, surgical correction could be achieved through extensive plantar release and metatarsal osteotomies. However, at the time of the patient’s first visit, physicians from the Yucatan Project prioritized the management of the ulcer prior to correcting any gross deformity. Due to the long period of treatment for ulcer management and limited medical access in the Yucatan Peninsula, the patient did not seek medical help for many years. Recent literature has described that if left untreated, cavovarus deformity can progress into fibrosis of the plantar fascia, shortening and tightening of the achilles tendon leading to excess pressure under metatarsal heads, overloading of the lateral aspect of the foot leading to stress fractures of the 5th metatarsal and more rarely, the cuboid. In addition, it can cause inadequacy of the lateral ligaments and tendons leading to instability of anterolateral ankle and lateral talus [15]. During the 2020 Covid-19 pandemic, we reached out through social media and discovered the patient was no longer ambulating. The patient described a rigid deformity with insensate feet and showed interest in undergoing another surgery, so she could possibly walk again. In a mature foot, surgical intervention might require aggressive techniques including midtarsal osteotomies, calcaneal osteotomies and triple arthrodesis [16]. Final decision for a surgical procedure will only be done in person once full updated history and radiographs are taken.

Case 3 showed the most severe result that could come from insensate lower extremity in spina bifida patients if left untreated for long periods of time: amputation. This patient was first seen at 24 years old, when his rigid cavovarus deformity was present since birth. This deformity caused chronic non-healing wounds that developed into osteomyelitis. Osteomyelitis can be defined as an infectious agent which causes inflammation of the bone. The hallmark of chronic osteomyelitis is the progression of inflammation to tissue necrosis and destruction of bone trabeculae and bone matrix caused by an infectious agent. This is usually accompanied by fragments of bone lacking blood supply which can become separated to form sequestra and continues to host and spread bacteria despite antibiotic treatment. The fifth metatarsal, first metatarsal, calcaneus, and first digit distal phalanx are the four structures with the highest incidence of developing osteomyelitis in the foot [17]. This case emphasizes the need of spina bifida patients with concomitant lower extremity deformities to seek medical help at a young age to avoid the progression of the deformity and consequently loss of a limb.

Given that this study was built from reports of medical mission trips that occur four times a year with limited resources and time, the lack of documentation of treatment plans and follow-ups made it difficult to identify surgical procedures and assess the success of surgeries. Additionally, we did not have access to the patients birth records or their mothers medical records to accurately determine the etiology of their deformities.

Conclusion

The types of foot and ankle deformities seen in spina bifida are diverse in etiology, age and gender of the patients. We discovered the most common lower extremity manifestations of spina bifida in the Yucatan Peninsula are flexible equinus and rigid pes cavus. The mean age of patients with rigid deformities was almost twice as the mean age of the patients with flexible deformities. Zang, et al., concluded that equinovarus requires immediate treatment while valgus deformities can have delayed intervention [15]. Based on our data, we conclude that intervention should be considered as early as possible in any flexible deformity to prevent them from becoming rigid.

Acknowledgements

We would like to thank all attendings, residents and students involved in the Yucatan Crippled Children Project along with the International Foot & Ankle Foundation for Education and Research. Additionally, we would like to thank the local Red Cross Hospital in the city of Merida.

References

  1. Gunay H, Sozbilen MC, Gurbuz Y, Altinisik M, Buyukata B. Incidence and type of foot deformities in patients with spina bifida according to level of lesion. Childs Nerv Syst. 2016;32(2):315-319. doi:10.1007/s00381-015-2944-7.
  2. Swaroop VT, Dias L. Orthopaedic management of spina bifida-part II: foot and ankle deformities. J Child Orthop. 2011;5(6):403-414. doi:10.1007/s11832-011-0368-9.
  3. Sharrard WJ, Grosfield I. The management of deformity and paralysis of the foot in myelomeningocele. J Bone Joint Surg Br. 1968;50(3):456-465.
  4. Atta CA, Fiest KM, Frolkis AD, et al. Global Birth Prevalence of Spina Bifida by Folic Acid Fortification Status: A Systematic Review and Meta-Analysis. Am J Public Health. 2016;106(1):e24-e34. doi:10.2105/AJPH.2015.302902.
  5. Frischhut B, Stöckl B, Landauer F, Krismer M, Menardi G. Foot deformities in adolescents and young adults with spina bifida. J Pediatr Orthop B. 2000;9(3):161-169. doi:10.1097/01202412-200006000-00005.
  6. Krähenbühl N, Weinberg MW. Anatomy and Biomechanics of Cavovarus Deformity. Foot Ankle Clin. 2019;24(2):173-181. doi:10.1016/j.fcl.2019.02.001.
  7. Westcott MA, Dynes MC, Remer EM, Donaldson JS, Dias LS. Congenital and acquired orthopedic abnormalities in patients with myelomeningocele. Radiographics. 1992;12(6):1155-1173. doi:10.1148/radiographics.12.6.1439018.
  8. Yucatán Crippled Children’s Project. (2012). Retrieved from (https://www.internationalfootankle.org/philanthropy/crippled-childrens-project/).
  9. Mandell JC, Khurana B, Smith JT, Czuczman GJ, Ghazikhanian V, Smith SE. Osteomyelitis of the lower extremity: pathophysiology, imaging, and classification, with an emphasis on diabetic foot infection. Emerg Radiol. 2018;25(2):175-188. doi:10.1007/s10140-017-1564-9.
  10. Cavalheiro S, da Costa MDS, Moron AF, Leonard J. Comparison of Prenatal and Postnatal Management of Patients with Myelomeningocele. Neurosurg Clin N Am. 2017;28(3):439-448. doi:10.1016/j.nec.2017.02.005.
  11. McCluskey WP, Lovell WW, Cummings RJ. The cavovarus foot deformity. Etiology and management. Clin Orthop Relat Res. 1989;(247):27-37.
  12. Feldkamp, M., Sanchez, E., & Canfield, M. (2014). International Clearinghouse of Birth Defects Surveillance and Research – Annual Report 2014.
  13. Awang M, Sulaiman AR, Munajat I, Fazliq ME. Influence of Age, Weight, and Pirani Score on the Number of Castings in the Early Phase of Clubfoot Treatment using Ponseti Method. Malays J Med Sci. 2014;21(2):40-43.
  14. Copp AJ, Adzick NS, Chitty LS, Fletcher JM, Holmbeck GN, Shaw GM. Spina bifida. Nat Rev Dis Primers. 2015;1:15007. Published 2015 Apr 30. doi:10.1038/nrdp.2015.7.
  15. Zang J., Qin S., Shi L. (2020) Lower Limb Deformity Caused by Spina Bifida Sequelae and Tethered Cord Syndrome. In: Qin S., Zang J., Jiao S., Pan Q. (eds) Lower Limb Deformities. Springer, Singapore.
  16. Kancherla V. Countries with an immediate potential for primary prevention of spina bifida and anencephaly: Mandatory fortification of wheat flour with folic acid. Birth Defects Res. 2018;110(11):956-965. doi:10.1002/bdr2.1222.
  17. Broughton NS, Graham G, Menelaus MB. The high incidence of foot deformity in patients with high-level spina bifida. J Bone Joint Surg Br. 1994;76(4):548-550.

 

Management of a dislocated talar dome fracture with ankle arthrodiastasis and open reduction internal fixation: A case report

by Charles A. Sisovsky, DPM, AACFAS1*; Carl A. Kihm, DPM, FACFAS2

The Foot and Ankle Online Journal 13 (4): 7

Osteochondral lesions of the talus (OLT) can be acute or chronic with mechanisms of injury and treatment protocols that have been well-described. Current treatment options for OLT depend on severity and chronicity. Treatment options for OLT consist of bracing, steroid injections, arthroscopic debridement with microfracture, osteochondral transfer, structural allograft, arthrodiastasis, arthrodesis or total ankle arthroplasty. Although mechanisms are similar, talar dome fractures have been less frequently presented in our literature. Displaced intra-articular fractures often require operative management although these procedures have not been detailed in the literature due to the rarity of the injury. This case report describes the surgical management of a 19-year-old male who sustained a dislocated and rotated lateral talar dome fracture after an inversion ankle injury while playing basketball. Long-term follow-up of our patient show an excellent, asymptomatic outcome without limitations at his 18-month follow-up visit.

Keywords: arthrodiastasis, external fixation, osteochondritis, syndesmosis, talus fracture, transchondral fracture

ISSN 1941-6806
doi: 10.3827/faoj.2020.1304.0007

1 – Fellowship-trained Foot and Ankle Surgeon, American Health Network, OptumCare, Department of Foot and Ankle Surgery, 2108 State Street, New Albany, IN 47150
2 – Attending surgeon at Norton Audubon Hospital, Faculty of the Kentucky Podiatric Residency Program, Private Practice, 3 Audubon Plaza Drive Suite 320, Louisville, KY 40217
* – Corresponding author: csisovsk@gmail.com


Osteochondral lesions of the talus (OLT) have been thoroughly discussed throughout the literature and describe pathologies to include transchondral lesions, osteochondral lesions, and talar dome fractures. These lesions typically involve the talar cartilage and subchondral bone and are typically caused by a single or multiple traumatic events, leading to partial or complete detachment of the fragment [1]. These fractures comprise approximately 0.1-0.85% of all fractures and most occur as a result of high-energy trauma, such as motor vehicle accidents [2-3]. Talar dome lesions or osteochondritis dissecans were first described by Berndt and Harty in the ankle in 1959 [4]. Current treatment options for OLT range from non-surgical treatment with cast immobilization to surgical excision and microfracturing. Newer techniques include osteochondral autograft transplantation and autologous chondrocyte implantation. The goal of these treatments is to restore the anatomic alignment of the articular surfaces in order to diminish long-term pain and swelling, and to improve function. The surgical management of large, displaced talar dome fractures is difficult in that with the tight joint confines, anatomic reduction may require ankle joint distraction to achieve proper reduction. Both arthroscopic and open exposures are considered regardless of whether ankle arthrodiastasis is utilized. Then, once reduced, optimal fixation placement is also challenging due to the tight joint confines and since fixation must not be detrimental to the articular surfaces or prominent or impinge the ankle joint.

In this case, we present the surgical management we utilized to achieve open reduction internal fixation of a dislocated lateral talar dome fracture with external fixation arthrodiastasis. Since anatomic reduction was achieved and our patient had an excellent long-term outcome, here, we present the surgical technique employed which may be useful in similar, future displaced lateral talar dome cases as these are rarely described in our literature [5-6]. Our case is unique in that we described the surgical management of a completely dislocated talar dome fracture with the use of bioabsorbable pins and ankle joint distraction.

Case Report

A healthy 19-year-old male inverted his ankle while playing basketball, when landing after jumping. Immediately afterwards, he was unable to bear weight. He went to the emergency department where radiographs demonstrated a dislocated lateral talar dome fracture (Figure 1). A CT scan was ordered which confirmed that the fracture segment, which measured 2.2 x 1.3 x 0.6 cm, was dislocated and rotated 180 degrees (Figure 2). The patient was made non-weight bearing while soft tissues were managed for 2 weeks.

Surgical management of the lateral talar dome fracture dislocation started with application of a delta frame external fixation construct to manually distract the ankle joint and maintain distraction during the postoperative period. The tibial tuberosity was palpated and four fingerbreadths were measured and this was the entry point of the first trans-tibial pin (Arthrex, Naples, FL). A 1 cm longitudinal skin incision was made using a #15 blade. Blunt dissection was then carried down to the level of bone using a curved hemostat. Next, the tibia was pre-drilled and a 5.0 mm tibial Schanz pin was inserted using a T-handle. The multi-pin clamp was then applied to the tibial Schanz pin and used as a drill guide to insert the second tibial Schanz pin which was inserted next. Utilizing fluoroscopic imaging, the trans-tibial pins were noted to be bi-cortical and of appropriate length and orientation. Next, attention was directed to the medial aspect of the calcaneus where 1 cm longitudinal skin incision was made using a #15 blade.

IMG_2339 2.png

Figure 1 Preoperative radiograph showing a completely displaced talar dome fracture.

Blunt dissection was carried down to the level of bone using a curved hemostat. Utilizing fluoroscopic imaging, a 6.0 mm trans-calcaneal pin was then inserted. The tibial and calcaneal clamps were then connected with carbon fiber bars. While the ankle was maximally distracted, with manual distraction of the transcalcaneal pin, the assistant surgeon locked the construct into place.

Next, an open incisional approach allowed for visualization, reduction and fracture stabilization. We created a 10 cm longitudinal skin incision over the anterolateral ankle gutter. The incision started 2 cm proximal to the syndesmosis, traversing the ankle joint, and curving medially over the lateral border of the talus. As expected, the superficial peroneal nerve and its terminal divisions were encountered and subsequently protected throughout the entirety of the case. Next, the deep fascia was incised utilizing dissection scissors. The extensor digitorum longus tendon was retracted laterally and a capsular incision was made into the ankle joint, exposing the displaced talar dome fracture fragment. The fragment was then excised and prepared for reinsertion (Figure 3).

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Figure 2 CT scan showing dislocated talar dome fracture.

Fracture hematoma and any loose, overhanging soft tissues were excised. The ankle was then flushed with copious amounts of sterile saline. The fragment was reinserted into the ankle joint, and delicately placed in its anatomical position. Two 18 mm x 1.3 mm bioabsorbable poly-L-lactic acid Chondral Darts (Arthrex, Naples, FL) were then inserted in the anterior and posterior aspects of the fracture. With the limited access in the ankle joint, the pins were still placed in a diverging manner to provide greater capture and stability (Figure 4).

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Figure 3 Excised fragment measuring approximately 2.5 cm x 1.5 cm.

IMG_1490.png

Figure 4 Fragment re-implanted and fixated with bioabsorbable pins.

Intraoperative imaging confirmed clinical findings that the fracture was reduced and the talar dome restored.

Preoperative radiographs were suspicious for a distal tibiofibular diastasis which could not be ruled out given the rotational mechanism of injury. We tested the stability of the syndesmosis intra-operatively utilizing the Cotton hook test [7].

IMG_3161.jpg

IMG_2332.png

Figure 5 Successful application of suture button and external fixator.

This was done by applying a laterally-directed force on the fibula with a towel clamp which resulted in lateral translation of the fibula with respect to the tibia. Therefore, we made an intraoperative decision to transfix the syndesmosis. First, a periarticular clamp was placed perpendicular to the axis of the ankle joint, reducing the syndesmosis which was confirmed on fluoroscopy. Next, the syndesmosis was stabilized using knotless, trans-syndesmotic Tightrope® fixation (Arthrex, Naples, Florida).

IMG_2333.png

Figure 6 Radiograph 12 weeks postoperative.

Figure 7 Radiograph 16 weeks postoperative.

Final images were taken and the ankle was noted to be maintained in distraction, the syndesmosis reduced with the Suture Button system applied appropriately and the fragment restored to anatomic position (Figure 5). The incision was closed with vicryl sutures and stability was maintained through the external fixation.

He went on to heal uneventfully, without soft tissue complications, paresthesias or limitations in range of motion. The external fixator was removed after 6 weeks. Immediately thereafter, the patient began home physical therapy with ankle strengthening and range of motion exercises. By postoperative week 9, he started placing more pressure on his foot, partial-weight bearing in a pneumatic boot as he transitioned off crutches. By 12 weeks, he was completely off crutches and partial weight bearing in his boot.

Radiographs demonstrated progression of bone consolidation and maintained alignment (Figure 6). At 16 weeks he was back in his shoes and without pain or limitations. Radiographs appear normal but bony exostoses were beginning to form at the medial malleolus and medial talus.

There were no restrictions of motion upon physical exam (Figure 7). At the 18-month follow-up he stated he had been back to his regular daily activities, including basketball, with no reports of pain. Radiographs show excellent restoration of the talar dome and ankle mortise. Bony exostoses are noted but incidental and asymptomatic (Figure 8). According to the AOFAS Ankle-Hindfoot Scale (AHS) the patient scored 100 points and the patient is very pleased with his outcome, lack of pain and level of function.

Discussion

Osteochondral lesions (OCLs) of the talus are rare in terms of overall fracture type in the lower extremity, however, they can be present in more ways than one might think. Lambers et al. retrospectively reviewed data of a prospective cohort of 59 patients and showed the prevalence of OCLs of the talus in ankle fractures with syndesmotic instability was 14% with most lesions located on the lateral talar dome [8]. This is consistent with the mechanism of injury and location of fracture of the case presented. Conversely, Raikin, et al., reviewed 428 ankle MRIs and found that the medial talar dome was most involved 62% of the time and, of those, the medial and mid zone was affected 53% [9]. Whereas these lesions are typically more superficial, the case presentation here focused on a dislocated, larger fracture segment of the lateral talar dome and this has not been explained much in our literature.

Figure 8 Radiographs one year postoperative.

Diagnosis of OCLs may be elusive during the early stages of patient complaints and can result in a delayed diagnosis [10]. In our case, the patient had a completely detached and 180-degree rotated large fracture so diagnosis was clear and straightforward. In addition to the standard preoperative radiographs, we ordered a CT scan to get a more accurate depiction of the lesion. CT scans can help identify the amount of bone involvement in an OCL and help in determining ideal fixation methods [10].

Multiple treatment options are available for OCLs of the talus and arthrodiastasis is among the preferred options. Arthrodiastasis has been shown to benefit patients significantly in the short- and long-term in prevention of post-traumatic ankle arthritis. However, ankle arthrodiastasis is considered a salvage procedure and most often a last-ditch effort prior to fusion or ankle implant arthroplasty [11-12]. Although the present case did not have a patient with ankle arthritis, we used this option to slow the progression of future osteoarthritis.

Joint distraction is not only used in the ankle, it is also used in the hip and knee with good long-term outcomes [13-14]. Furthermore, joint distraction is also used in other aspects of the foot and ankle. Dayton, et al., published a review describing a percutaneous technique for calcaneal fractures. They concluded that their patients’ return to function was similar or better than after open reduction, and their soft tissue complication rate was much lower [15]. In ankle joint distraction, it’s recommended to obtain at least 5 mm of distraction [11]. We achieved 2.7 mm of joint distraction which was measured from the calibrated from the postoperative radiographs via the PACS program.

Arthrodiastasis is based on the theory that osteoarthritic cartilage has healing capacity. The chondrocyte repair is nourished by intra-articular fluid pressure changes within the joint by movement with the use of hinges in the external fixator or by allowing the patient to walk with the frame in place. This allows intermittent increases in hydrostatic pressure, creating a supportive environment for cartilage repair [11]. In our case we used a static external fixator and made the patient non-weight bearing due to the fact that the patient had a displaced talus fracture that was reduced using open reduction internal fixation. The arthrodiastasis effect is difficult to evaluate individually. The authors believe the external fixation provided added benefit of maximal ankle immobilization as the bioabsorbable pins maintained position but did not affect much compression or maximum fracture stability.

In conclusion, our case report describes the use of open reduction and internal fixation of a large, completely dislocated lateral talar dome fracture fixated with bioabsorbable fixation and further stabilized with external fixation ankle arthrodiastasis. Valderrabano, et al., completed a study of 390 patients and found that talus fractures accounted for 2% of patients acquiring post traumatic ankle arthritis [17]. Lastly, Nakasa, et al., showed favorable outcomes utilizing bioabsorbable PLLA pins even in those patients who had disruption of the subchondral plate (18). We supplemented our fixation with a delta frame in order to provide the patient with greater stability and to allow for ankle joint distraction. We felt that this was appropriate for a patient of his age to slow the progression of ankle osteoarthritis. At 1.5 years postoperative assessment, our patient is without pain, functioning without limitation and pleased. This treatment approach may be beneficial in patients presenting with similar pathology. Further research investigating the risk of post traumatic ankle arthritis is needed to better understand long-term outcomes with this procedure.

References

  1. Zenegrink M, Struijs PA, Tol J, van Dijk C. Treatment of Osteochondral Lesions of the Talus: A Systematic Review. Knee Surg, Sports Traumat, Arthroscopy 8(2):238-246, 2010.
  2. Fortin, P. and Balazsy, J. Talus Fractures: Evaluation and Treatment. Journal American Acad of Ortho Surgeons 9(2):114-127, 2001.
  3. Higgins, T. and Baumgaertner, M. Diagnosis and Treatment of Fractures of the Talus: A Comprehensive Review of the Literature. Foot Ankle Int 20(9):595-605, 1999.
  4. Berndt, A. and Harty, M. Transchondral Fractures (Osteochondritis Dissecans) of the Talus. The J Bone Joint Surg Am 41(7):988-1020, 1959.
  5. D’Angelantonio AM, Schick FA. Ankle Distraction Arthroplasty Combined with Joint Resurfacing for Management of an Osteochondral Defect of the Talus and Concomitant Osteoarthritis: A Case Report. J Foot and Ankle Surg 52(1):76-79, 2013.
  6. Belczyk R, Stapleton JJ, Zgonis T, Polyzois VD. A Case Report of a Simultaneous Local Osteochondral Autografting and Ankle Arthrodiastasis for theTreatment of a Talar Dome Defect 26:335-342, 2009.
  7. Cotton FJ. Fractures and Joint Dislocations p 549. WB Saunders, Philadelphia, 1910.
  8. Lambers KA, Saarig A, Turner H, Stufkens SA. Prevalence of Osteochondral Lesions in Rotational Type Ankle Fractures With Syndesmotic Injury. Foot Ankle Int 40(2):159-166, 2019.
  9. Raikin SM, Elias I, Zoga AC, Morrison WB, Besser MP, Schweitzer ME. Osteochondral Lesions of the Talus: Localization and Morphologic Data from 424 Patients Using a Novel Anatomical Grid Scheme. Foot Ankle Int 28(2):154-61, 2007.
  10. Talusan PG, Milewski MD, Toy JO, Wall EJ. Osteochondritis Dissecans of the Talus: Diagnosis and Treatment in Athletes. Clin Sports Med 33:267-284, 2014.
  11. Kluesner AJ, Wukich DK. Ankle Arthrodiastasis. Clin Podiatr Med Surg 26:227-244, 2009.
  12. Labovitz JM. The Role of Arthrodiastasis in Salvaging Arthritic Ankles. Foot Ankle Spec 3(4):201-204, 2010.
  13. Hosny GA, El-Deeb K, Fadel M, Laklouk M. Arthrodiastasis of the Hip, J Pediatr Orthop 31(2):S229-234, 2011.
  14. Jansen MP, Besselink NJ, van Heerwaarden RJ, Custer RJ, Emans PJ, Spruijt S, Mastbergen SC, Lafeber FP. Knee Joint Distraction Compared with High Tibial Osteotomy and Total Knee Arthroplasty: Two-Year Clinical, Radiographic, and Biochemical Outcomes of Two Randomized Controlled Trials. Cartilage, 2019.
  15. Dayton P, Feilmeier M, Hensley NL. Technique for minimally invasive reduction of calcaneal fractures using small bilateral external fixation. J Foot Ankle Surg. 53(3):376–82, 2014.
  16. Valderrabano V, Horisberger M, Russell I, Dougall H, Hintermann B. Etiology of Ankle Osteoarthritis. Clin Orthop Relat Res 467(7): 1800-1806, 2009.
  17. van Valburg AA, van Roermund PM, Lammens J, van Melkebeek J, Verbout AJ, Lafeber EP, et al. Can Ilizarov joint distraction delay the need for an arthrodesis of the ankle? A preliminary report. J Bone Joint Surg Br. 77(5):720–5, 1995.
  18. Nakasa T, Ikuta Y, Tsuyuguchi Y, Ota Y, Kanemitsu M, Adachi N. Fixation Technique Using PLLA Pins Gives Good Clinical Results Regardless of Bone Condition in Osteochondral Lesion of Talar Dome. Foot & Ankle Orthopaedics. 2019 Oct 1;4(4):2473011419S0031.

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A case of recurrent hyperostotic macrodactyly

by Milad Motalebi Kashani DPM1*, Melinda A. Bowlby DPM2

The Foot and Ankle Online Journal 13 (4): 6

Macrodactyly and its variation hyperostotic macrodactyly are some of the rarest deformities encountered by foot and ankle specialists. Changing the natural aesthetic shape of the foot, limiting the functionality of the lower extremity, and causing pain are some ways that this condition can affect patients’ everyday life and mental health. This study presents a case of recurrent hyperostotic macrodactyly that was managed with surgical intervention in order to debulk the soft tissue and excise excess osseous elements with successful results.

Keywords: congenital deformity, forefoot, lower extremity, foot and ankle surgery

ISSN 1941-6806
doi: 10.3827/faoj.2020.1304.0006

1 – Swedish Medical Center- Cherry Hill Campus, Seattle, WA PGY-1
2 – Assistant Research Director, Swedish Medical Center-Cherry Hill Campus, Seattle, WA
* – Corresponding author- miladm14@vt.edu


Macrodactyly is a non-hereditary and congenital deformation pathology of the upper and lower extremities which can be bilateral or unilateral [1-5]. In this deformity both osseous and soft tissue components of the digit can be enlarged in size which can cause functional and esthetic problems for the patient. Two types of this condition that were described by Barsky are static, in which deformity is present at birth and increases in size proportionally to other digits, and progressive, in which a digit grows disproportionately to other digits and is most commonly encountered in the lower extremity [1].

Macrodactyly is believed to be originally described by Von Klein in 1824 in the upper extremity and later in 1925 by Feriz in the lower extremity [6-8]. This disorder is thought to be a rare condition and because of that there is no accurate estimate of the prevalence of this disorder. According to some estimates, macrodactyly accounts for 0.9% of all congenital deformities and it is more prevalent in males [9, 10]. The etiology of macrodactyly has been debated over the years, but no clear conclusion has emerged yet. Some cases of macrodactyly present in patients with other disorders such as Proteus syndrome, Neurofibromatosis type 1, Klippel–Trenaunay syndrome, lymphangioma and fibrous dysplasia [8,11,12]. However, in many of the case reports published regarding macrodactyly, this condition is observed as an isolated condition with no other associated disorders [2,3,9,13,14].

Throughout the years, different terms such as macrodystrophia lipomatosa progresia, macrodystrophia lipomatosa, megalodactyly, and localized gigantism have been used in the literature to describe macrodactyly or other variations of this deformity [7]. A unique and less discussed type of progressive macrodactyly is hyperostotic macrodactyly which usually has a later onset than typical macrodactyly and is associated with osteo-cartilaginous mass formation in peri-articular areas of the upper and lower extremity [15].

Figure 1 Preoperative medial oblique X-ray image (on the left) and clinical photo (on the right), prior to second surgery.

Figure 2 Postoperative medial oblique X-ray image, following the second surgery.

The rare presentation of hyperostotic macrodactyly and a lack of literature regarding this topic have provided practitioners with no clear guidelines regarding the management of this disorder. This case report presents a case of a mild recurrent hyperostotic macrodactyly in a patient with previous surgical interventions to address this deformity.

Case Report

A 53-year-old female with no past medical history other than asthma presented to the clinic with a painful recurrent mass on her left hallux. She related that she has had two prior surgeries. The patient reported that she was originally seen regarding this problem when she was 14 years old and was diagnosed with localized gigantism. She had a surgery at that time to fix her deformity which had satisfying results and resolved her problem for about 35 years.

The second surgery was performed four years ago when the patient was seen by another podiatrist regarding this problem. She had noticed an increase in size of her left hallux and denied any trauma to the area. She had pain both on the plantar and medial side of her left hallux and first metatarsal head with noticeable bony prominences. She changed her shoe gear, using slippers or open sandals to accommodate the prominences (Figure 1). In the second surgery, osteophytes from the left first metatarsophalangeal joint and hallux were excised and a soft tissue mass from the plantar aspect of the left hallux was removed as well (Figure 2). During the postoperative period, the patient stubbed her hallux on a heavy plastic bin which was very painful for her, however no fractures were noted, and the rest of her postoperative course was uneventful.

The patient presented to our clinic two years ago due to noticing the recurrence of her deformity after the second surgery. Physical exam revealed approximately a 4 cm x 3 cm firm soft tissue mass overlying the left first tarsometatarsal joint. There was also tenderness with palpation of a prominent exostosis along the medial aspect of the left hallux Interphalangeal joint. Joint motion at first metatarsophalangeal and tarsometatarsal joints were severely limited as well, but not painful (Figure 3). Magnetic resonance imaging report indicated an ovoid, subcutaneous lipoma measuring 5.2 x 2.8 x 1.1 cm. Mild to severe arthritis of the first tarsometatarsal and metatarsophalangeal joints with ossified bodies in addition to fatty infiltration of abductor and flexor hallucis muscles was also noted.

Figure 3 Preoperative clinical photos (in the left and the middle) and dorsoplantar X-ray image (on the right), prior to the third surgery.

After discussing the possible adverse effects, benefits and alternative therapies to the surgery with the patient, the patient wished to proceed with exostectomy and removal of the soft tissue mass. During the third surgery, osseous masses from the left hallux were removed and the soft tissue mass from the dorsal and medial aspect of first metatarsal of the left foot were excised and both specimens were submitted for pathology evaluation (Figure 4). The osseous masses were clinically equivalent with osteophytes measuring 0.8×0.6×0.5cm and 1.2×0.8.0.5cm.

Figure 4 Intraoperative image of the excised fibro-fatty mass, during the third surgery.

Figure 5 Postoperative clinical photo, following the third surgery.

The soft tissue mass revealed mature adipose tissue with features of a lipoma measuring 4.5×2.5×1.3cm. The patient was kept non-weight bearing in a splint for 2 weeks and then she was allowed to weight-bear as tolerated for 2 weeks in a surgical boot. Postoperative course was uneventful, and the patient made good progress, had no complaints, and was satisfied with the results more than 18 months after the third surgery (Figure 5).

Discussion

Hyperostotic macrodactyly is a distinctive type of macrodactyly in which massive osteo cartilaginous deposits are observed around the joints [2,15]. Early in the formation process of these osteo-cartilaginous bodies around the joints, they are mostly cartilaginous and later they are substituted with osseous elements which leads to motion restriction across the affected joint [2]. As a progressive macrodactyly, it is not uncommon to observe fatty growths or lipomas and fatty infiltrations in this condition. In many reported cases this fatty hypertrophy and infiltration can be observed in both plantar and dorsal aspect of the foot [3,14]. The case presented in this article demonstrated both osseous and fatty enlargement across the first ray of the patient’s left foot.

The main goals of treatment for macrodactyly should be providing the patients with a pain free, functional foot that can fit into their shoes [4]. Unfortunately, there are very few reported cases of hyperostotic macrodactyly in the foot, however it appears that the removal and debulking of the excess fibro-fatty tissue and osseous bodies is the best method of management of this disorder which was the way the patient in this case was treated [2,9,13]. In a case report by Katz, the patient presented with lipomas across his right foot and ankle and osteo-cartilaginous growths across his fourth and fifth toes and metatarsals [9]. An surgery was performed on the patient in which the lipomas and osteo-cartilaginous bodies were excised and the fifth toe and half of the fifth metatarsal were resected. The patient had excellent results after the surgery. In another case by Matsuzaki, et al., the patient was suffering from painful and limiting osteo-cartilaginous masses around his left first metatarsal head and ankle [2]. Patient had a previous left second toe and hallux amputation surgery to address his macrodactyly. After removal of these osseous bodies from the patient’s left foot, his pain was relieved and the motion across his ankle joint increased and no recurrence was reported.

One curious aspect of the case presented in this article that requires attention is the recurrence of osteo-cartilaginous bodies almost two years after their resection in the second operation. This seems to be too early for a recurrence to happen considering that it took almost 35 years after the first surgery for her to have problems with her left foot again. A possible explanation for this can be the traumatic event to the area in the postoperative course in which the patient stubbed her big toe straight into a heavy plastic bin. Some studies have suggested that trauma can be the trigger for the osteo-cartilaginous hypertrophy observed in hyperostotic macrodactyly deformity which would explain the recurrence in this case [2,15].

In conclusion, hyperostotic macrodactyly is a rare progressive form of macrodactyly in which massive and limiting periarticular osteo-cartilaginous bodies in addition to fatty tissue hypertrophy can form. Surgical intervention to remove these osseous and fatty masses in cases that they cause pain and functional disability due to blocking joints appears to be the best method to treat this condition. It is also very important to educate patients to avoid trauma to the areas affected by hyperostotic macrodactyly since trauma appears to be one of the causes of this disorder or its recurrence.

References

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