Category Archives: Uncategorized

Navicular dislocation and orthotic management: A case study

by Joshua Young BSc.(Hons), MBAPO Orthotist1*

The Foot and Ankle Online Journal 11 (3): 2

Navicular dislocation is a rare injury, typically managed by surgical fixation. This case study presents the results of conservative management of navicular dislocation, using a custom foot orthosis, combined with a removable walker boot. At 6 week review the numeric pain rating scale (NRS-11) score was reduced from 8/10 to 3/10. A foot orthosis combined with a removable walker boot may reduce pain in the short term in non-operable navicular dislocation more successfully than a walker boot alone.

Keywords: navicular, navicular dislocation, orthoses, orthotics

ISSN 1941-6806
doi: 10.3827/faoj.2018.1103.0002

1 – Roehampton Rehabilitation Centre, Queen Mary’s Hospital. St George’s University Hospitals NHS Foundation Trust; Opcare, Oxfordshire, UK.
* – Corresponding author: Joshua.Young@stgeorges.nhs.uk


Dislocation of the navicular without fracture is a rare injury [1-3]. A 2015 case study by Singh and colleagues found 16 previously reported cases in the literature [2].  A further case study was published in 2016 [1]. Two other published cases were not identified which gives a total estimate of 20 published cases in the literature to date [4,5].

The mechanics of injury are commonly described as involving pronation, with abduction of the forefoot [6]. Davis and colleagues describe a transient midtarsal dislocation which allows the navicular to dislocate [3]. The navicular may displace in a plantar or dorsal direction, depending on the nature of the injury.  Dhillon and Nagi argue that the injury is never truly an isolated injury as disruption to both the medial and lateral columns of the foot is necessary [6].

Surgical management is usually recommended, typically with temporary Kirschner wire fixation, although other means of fixation may be used [1-3].  Custom foot orthoses have been suggested as a possible treatment option for surgically corrected cases of navicular dislocation which remain painful  however there are no reported cases of purely conservative management of navicular dislocation of which the author is aware [1]. This case report presents a navicular dislocation managed purely conservatively using orthoses due to surgical risk factors which made the subject a poor candidate for surgery.

Case report

A 67-year-old male was referred to see an orthotist in the orthotic service by his orthopedic foot and ankle consultant. The subject had an injury to his left foot 5 months previously sustained during a fall which occurred whilst walking indoors. Initial radiographs and computed tomography scans following the fall show a dislocated navicular and cuboid fracture (Figures 1-4). One month post injury radiographs showed reduced 11 degree calcaneal inclination angle on the left (affected) side compared to 24 degrees on the right, reflecting a reduction in height of the medial longitudinal arch.

Figure 1 Dislocated navicular resting inferior to the sustentaculum tali.

Figure 2 Dislocated navicular resting inferior to the sustentaculum tali.

Figure 3 Cuboid fracture.

Figure 4 Three dimensional CT reconstruction showing dislocated navicular.

A significant factor in the injury was the subject’s body weight which at 2 months following the injury was 161kg (Body Mass Index 50). At the time of assessment in orthotic clinic 3 months later, this had increased to 189kg (Body Mass Index 56). The increased body weight will have increased the ground reaction forces experienced by the foot, and the resultant internal stresses on tissues such as ligamentous tissue which normally help to maintain joint congruency [7,8].

Following two orthopedic opinions, and assessment in a high risk anesthetic clinic, it was agreed to avoid surgery due to the high risk of mortality. It was observed that the talar head was now articulating with the medial cuneiform, forming a pseudo joint.

At presentation in orthotic clinic the subject reported pain as the primary concern. His walking was very limited to short distances indoors, wearing a removable walker boot (Aircast Airselect, Donjoy). He reported pain at an intensity of up to 8 out of 10 (numeric rating scale, NRS-11). A custom ankle foot orthosis (AFO) was considered to limit painful movement within the foot, however this was decided to not be feasible as the subject would struggle to apply or remove this independently [9]. A custom foot orthosis (FO) was prescribed to wear inside the walker boot. The mechanical aim of this was to apply forces to the medial longitudinal arch in an attempt to modify compressive stresses assumed to be occurring at the midfoot and talo-cuneiform pseudo-joint, and tensile stresses assumed to be occurring in soft tissues at the plantar foot [9]. The FO was made from an imprint of the foot in a foam impression box using a computer aided design and manufacture (CAD-CAM) system (Paromed, Neubeuern, Germany). The FO was manufactured using 70 shore material at the heel to midfoot, and softer 50 shore material from the midfoot to the forefoot. A soft 3.2mm grey poron polyurethane foam cover (Algeos, Liverpool, UK) was added. The shape of the FO is demonstrated by the modeling images (Figure 5a-d).

FAOJ_11.3.2 (1)

Figure 5 Custom foot orthosis, medial view (a), lateral view (b), anterior view (c), dorsoplantar view (d).

At 6 week review the subject reported good compliance with wearing the FO within the walker boot. Using the 11 point numeric pain rating scale (NRS-11), pain intensity during walking was reported to be reduced from 8/10 to 3/10.

Discussion

This case study presents the results of conservative management of an unusual foot injury. A custom FO, combined with a walker boot, reduced pain intensity in the short term. Pain was still present while using the walker boot only. The reported reduction in pain following addition of the FO may imply that the FO was able to modify stresses in the midfoot, even in the presence of very high body weight, in order to be effective. FOs are rarely used for this specific application due to the rarity of the injury, however they may be combined  with walker boots to manage Charcot foot which is also associated with major change of the midfoot architecture. Limitations of this study include a lack of further outcome measures, and possible bias incurred by the treating clinician administering the NRS-11 pain scale. This case study illustrates the possibility that even major changes in the bony structure of the foot, which are symptomatic, may be manageable to some extent conservatively using foot orthoses.

Acknowledgements

The author would like to acknowledge Dominic Nielsen, consultant orthopaedic surgeon, for his comments on the paper.

References

  1. Ansari MAQ. Isolated complete dislocation of the tarsal navicular without fracture: A rare injury. Ci Ji Yi Xue Za Zhi. 2016;28(3):128-131.
  2. Singh VK, Kashyap A, Vargaonkar G, Kumar R. An isolated dorso-medial dislocation of navicular bone: A case report. J Clin Orthop Trauma. 2015;6(1):36-8.
  3. Davis AT, Dann A, Kuldjanov D. Complete medial dislocation of the tarsal navicular without fracture: report of a rare injury. J Foot Ankle Surg. 2013;52(3):393-6.
  4. Hamdi K, Hazem BG, Yadh Z, Faouzi A. Isolated dorsal dislocation of the tarsal naviculum. Indian J Orthop. 2015;49(6):676-9.
  5. Dias MB, Zagonel B, Dickel MS, Talheimer JA, Argenton IS, et al. Isolated dislocation of the tarsal navicular without fracture: Case report. Trauma Cases. 2016,Rev 2:045
  6. Dhillon MS, Nagi ON. Total dislocations of the navicular: are they ever isolated injuries?. J Bone Joint Surg Br. 1999;81(5):881-5.
  7. Browning RC, Kram R. Effects of obesity on the biomechanics of walking at different speeds. Med Sci Sports Exerc. 2007;39(9):1632-41.
  8. Pamukoff DN, Lewek MD, Blackburn JT. Greater vertical loading rate in obese compared to normal weight young adults. Clin Biomech (Bristol, Avon). 2016;33:61-65.
  9. International standards organization. ISO 8549-3, Prosthetics and orthotics – Vocabulary – Part 3: Terms relating to external orthoses. 1989.

Intramedullary fixation of distal fibular fractures in a geriatric patient: A case report

by Amanda Kamery DPM1*, Craig Clifford DPM MHA FACFAS FACFAOM2

The Foot and Ankle Online Journal 11 (3): 1

Intramedullary rod fixation is presented as a viable treatment option for distal fibular fractures in the geriatric population. This technique leads to a reduction in wound complications, hardware irritation, procedure time and need for subsequent surgeries as seen with traditional open reduction internal fixation for distal fibular fractures in higher-risk patients.

Keywords: ankle fracture, trauma, geriatric, open reduction

ISSN 1941-6806
doi: 10.3827/faoj.2018.1103.0001

1 – Franciscan Foot and Ankle Institute- St Francis Hospital, Federal Way, WA PGY-3
2 – Residency Director, Franciscan Foot and Ankle Institute- St Francis Hospital, Federal Way, WA
* – Corresponding author: akamery@kent.edu


Geriatric patients are at an increased risk for sustaining ankle fractures due to increased fall rate and decreased bone density. Surgical repair for such injuries is often complex, due to the standard large incision and relatively bulky fixation which is necessary in the geriatric patient due to their generally poor bone stock [1]. This traditional form of fixation carries a complication rate of up to 30% [2]. Additionally, wound healing complications and hardware irritation is more common in this population due to a poor soft tissue envelope, with wound infection rates ranging from 26-40% [3]. Commonly, subsequent surgeries are necessary to remove hardware or to perform wound debridements [4]. As it is well documented that surgical morbidity increases in this population, it is important to utilize techniques and fixation methods that limit subsequent encounters. In this case report, we present intramedullary fixation for distal fibular fractures as a viable option for the geriatric population.

Case  Report

The patient is a 94-year-old male who presented 5 days after a fall with a Weber B, slightly comminuted, left distal fibular fracture (Figure 1a). Due to the unstable nature and slight displacement of the fracture, surgical intervention with an intramedullary fibular rod was chosen. Intra-operatively under general anesthesia, excellent anatomic reduction was noted after placement of the rod and one syndesmotic screw (Figure 1b).

At 2 weeks postoperatively, the posterior splint and skin staples were removed. The patient transitioned to protected heel touch weight-bearing for 4 weeks. He resumed regular activity and normal shoe wear at 6 weeks postoperatively. There were no wound healing complications or hardware irritation noted throughout the postoperative course. At 12 months follow up, patient reported no ankle pain or limitations in activities of daily living (Figures 2a-b).

 

Figure 1 AP ankle radiograph showing Weber B fracture with slight comminution and displacement (a). Two weeks postoperative AP radiograph showing excellent anatomic reduction with fibular rod and syndesmotic screw (b).

 

Figure 2 Twelve months post operative AP (a) and lateral (b) radiographs showing excellent bony consolidation of fracture fragments and adequate anatomic reduction.

Discussion

Treatment of distal fibular fractures in geriatric patients have an increased risk for postoperative complications which can lead to wound healing issues and subsequent surgeries. It is important to utilize techniques and fixation methods that limit subsequent encounters in order to decrease surgical morbidity in this cohort. The intramedullary fibular rod is an excellent alternative to traditional ORIF in the geriatric population. Our case example demonstrates an ideal patient for this technique, including successful anatomic realignment and uneventful postoperative course.

References

  1. Mitchell JJ, Bailey JR, Bozzio AE, Fader RR, Mauffrey C. Fixation of distal fibula fractures: an update. Foot Ankle Int. 2014;35(12):1367-1375.
  2. Lamontagne J, Blachut PA, Broekhuyse HM, O’Brien PJ, Meek RN. Surgical treatment of a displaced lateral malleolus fracture: the antiglide technique versus lateral plate fixation. J Orthop Trauma. 2002;16(7):498-502)
  3. Höiness P, Engebretsen L, Stromsoe K. The influence of perioperative soft tissue complications on the clinical outcome in surgically treated ankle fractures. Foot Ankle Int. 2001;22(8):642-648.
  4. Lee YS, Huang HL, Lo TY, Huang CR. Lateral fixation of AO type-B2 ankle fractures in the elderly: the Knowles pin versus the plate. Int Orthop 2007;31:817–821.

 

Summer 2018

Issue 11 (2), 2018


Progression of a digital or partial ray amputation to transmetatarsal amputation and below knee amputation: Time frames and associated comorbidities, a three-year retrospective study
by Carmen Bruno DPM, Susan Wiersema DPM, Nneka Meka DPM


The vacuum phenomenon in the ankle joint: Air bubbles on CT
by Christopher R. Hood JR. DPM, Wesley A. Jackson DPM, Robert C. Floros DPM, David A. Bernstein DPM


Operating on patients with complex regional pain syndrome
by Ryon Wiska DPM, Lawrence Fallat DPM FACFAS


Single lateral incision for a triple arthrodesis
by Alan Kidon DPM AACFAS, Elizabeth Sanders DPM AACFAS FACFAOM, Mark Mendeszoon DPM FACFAS FACFAOM


Single lateral incision for a triple arthrodesis

by Alan Kidon, DPM, AACFAS1; Elizabeth Sanders DPM, AACFAS, FACFAOM2*; Mark Mendeszoon DPM, FACFAS, FACFAOM3

The Foot and Ankle Online Journal 11 (2): 4

The triple arthrodesis surgical approach typically involves two incisions placed laterally and medially to obtain access to the subtalar joint (STJ), talonavicular joint (TN), and the calcaneocuboid joint (CC).  Despite the wide use of the two-incision approach, the traditional approach for triple arthrodesis has been described with a single lateral incision from the tip of the fibula across the sinus tarsi to the talonavicular joint, documented historically by Ollier.  The operative technique is described, 14 patients met the inclusion criteria, with a mean age of 50 (range 16-68). The most common diagnosis was posterior tibial tendon dysfunction (PTTD) (71%). Two (14.2%) required revisional operations: one developed a metal allergy and required hardware removal with soft tissue debridement and skin grafting all after achieving bony healing (7.1%), one developed a rearfoot varus requiring a dwyer calcaneal osteotomy (7.1%).  The union rate of each joint was 100% in the STJ, 100% in the TN joint, and 92.9% in the CC joint at 6 months post-operatively. The retrospective mean VAS pain score pre-operatively was 83, while the post-operative score was 42. The mean preoperative Talonavicular angle, Meary’s angle and Calcaneal pitch angle were 4.2; 4.8 and 15.2 respectively. The Immediate post-operative angles of each angle were 2.5; 1.2 and 22.5 respectively. At six months follow up, these angles were 2.6; 1.2 and 18 respectively.  

Keywords: triple arthrodesis, single lateral incision, exposure, union

ISSN 1941-6806
doi: 10.3827/faoj.2018.1102.0004

1 – Attending Surgeon, Ankle & Foot Care Centers, Boardman, OH
2 – Foot and Ankle Surgical Fellow, University Hospitals Richmond Medical Center, Precision Orthopaedic Specialties, Inc, Chardon, OH
3 – Attending Surgeon and Fellowship Director, Precision Orthopaedic Specialties, Inc, Chardon, OH
* – Corresponding author: elizabeth.sanders014@gmail.com


The triple arthrodesis is a widely accepted surgical method for treating complex rearfoot deformity and arthritis. The surgical approach typically involves two incisions placed laterally and medially in order to grant access to the subtalar joint (STJ), talonavicular joint (TN) and the calcaneocuboid joint (CC) [1]. Despite the wide use of the two-incision approach, the traditional approach for triple arthrodesis has been described with a single lateral incision from the tip of the fibula across the sinus tarsi to the talonavicular joint, documented historically by Ollier [2]. The triple arthrodesis is indicated in conditions such as post-traumatic arthritis, tarsal coalition, congenital deformities, neuromuscular deformities and end stage arthritis [3]. As the accepted gold standard for treating these problems, the procedure has since been modified and adapted in more recent years [4]. While adhering to the principles of fixation, modified approaches have been adapted which still allow for proper exposure and preparation of the joints. Appropriate alignment of the joints is paramount if a good result is to be achieved. The single incision approach has been described in literature from a single incision medial approach for triple arthrodesis and double arthrodesis with some success [5]. We hypothesized that correction would be able to be achieved and maintained through one lateral incision approach for triple arthrodesis. The aim of the retrospective study was to evaluate the complications and results of triple arthrodesis performed using a single lateral incision and to measure radiographic changes over a period of six months.

Methods  

Medical records were reviewed of all patients that underwent a triple arthrodesis with a single lateral incision, performed by one surgeon from January 2008 until February 2016.  Patients were excluded from the study if they had less than six months of follow up, if medical documentation was incomplete, and if external fixation was used during the initial surgery.  Data obtained retrospectively from chart review included patients’ age, gender, preoperative diagnosis, total surgical time, complications, and adjunctive surgeries that were performed. Pre- and postoperative radiographic measurements of talonavicular angle, Meary’s angle and calcaneal pitch angle were collected and calculated.  All surgeries were performed by one physician (MM). A preoperative and postoperative score for pain was obtained by VAS scale (Visual Analog Scale for Pain) via phone survey. Analysis was then conducted to calculate operative time.

Operative Technique

The patient is positioned on the operative table in the supine position with a pre-fashioned bump placed under the operative hip as deemed necessary.  A thigh tourniquet is placed. The single incision is then utilized from the distal portion of the fibula over the sinus tarsi to the base of the 4th metatarsal on the operative foot (Figure 1).   

Figure 1 Incision placement for the single lateral incision triple arthrodesis extending from just below the lateral malleolus extending across the sinus tarsi to the base of the 4th metatarsal.

Figure 2 Dissection and exposure for the single lateral incision for triple arthrodesis.

The sinus tarsi can be located by inserting a needle into the sinus tarsi prior to drawing out the incision.  At the distal most portion of the surgical approach, the extensor digitorum brevis is identified and split longitudinally to gain exposure to the calcaneocuboid joint.  Next, the interosseous ligament within the sinus tarsi is resected and any subcutaneous tissue is removed. Careful dissection is continued to level of the talonavicular joint, where a combination of elevators and positioning of the foot allows for release of capsular tissues (Figure 2).  Using curettes, round burr, and curved osteotomes; articular cartilage is carefully resection from the CC and ST joints.

Figure 3 Exposure of the talonavicular joint and subtalar joints visualized with a pin distractor through a single lateral incision.

Figure 4 Distraction and preparation of the calcaneocuboid joint with a pin distractor.

A cervical spine distractor is utilized to aid in preparation of these joints (Figures 3 and 4).  This process is then repeated to expose and prepare the TN joint, while carefully maintaining anatomical alignment of the joint.  The joints are then further prepared using a 2.0 mm drill bit to fenestrate each joint. Temporary fixation is utilized and permanent fixation is achieved with one or two 7.0 mm cannulated screws to fuse the STJ followed by two 5.5 mm screws to fuse the TN joint (Figure 5).  The CC joint is evaluated and fused using one or two staples.

Figure 5 Percutaneous fixation of the talonavicular joint.

Figure 6 Post-operative scars of two brothers, both with posterior facet tarsal coalitions, now status-post triple arthrodeses performed with the single lateral incision.

Closure is performed in layers.  A Jackson-Pratt drain is placed. The Silfverskiold test is performed after arthrodesis to evaluate the need for tendo-Achilles lengthening or gastrocnemius recession.  These procedures are performed concomitantly as deemed necessary by the surgeon.

Figure 7 Pre-operative and Post-operative films status-post triple arthrodesis through a single lateral incision.

The patient is placed into a posterior splint and Jones compression dressing for the first week after surgery.  At the first follow up visit at one week, the patient is placed into a below knee cast for three additional weeks.  At the fourth postoperative week, the cast is removed and the patient is placed into a removable walking boot. Over the course of week 5 and week 6, gradual weight bearing is increased until the patient is fully weight bearing in the boot by the end of the sixth week (Figures 6 and 7).    

Results

14 patients met the inclusion criteria, 5 males (35.7%) and 9 females (64.3%) with a mean age of 50 (range 16-68).  The average BMI of the group was 34.1(range 19.6-48.7). The most common diagnosis of the patients operated on in this study was posterior tibial tendon dysfunction (PTTD) (71%).  The mean operating time of the single incision triple arthrodesis was 90 minutes (range 60-135 minutes). Of the 14 patients included in this study, two (14.2%) required revisional operations.  One patient developed an unforeseen metal allergy and required hardware removal with soft tissue debridement and skin grafting all after achieving bony healing (7.1%). The other patient developed a postoperative rearfoot varus deformity and returned to the operating room for a dwyer calcaneal osteotomy (7.1%).  One patient (7.1%) developed a non-union of the CC joint which was asymptomatic. The union rate of each joint was 100% in the STJ, 100% in the TN joint, and 92.9% in the CC joint at 6 months post-operatively. The retrospective mean VAS pain score pre-operatively was 83, while the post-operative score was 42. The mean preoperative Talonavicular angle, Meary’s angle and Calcaneal pitch angle were 4.2; 4.8 and 15.2 respectively.  The Immediate post-operative angles of each angle were 2.5; 1.2 and 22.5 respectively. At six months follow up, these angles were 2.6; 1.2 and 18 respectively. After analysis using the unpaired t-test, the P-values were demonstrated in table 4. Values of less than 5% (p < 0.05) were considered statistically significant. All surgical corrections as measured with the three angles listed were found to be statistically significant in the immediate post-operative period.  At 6-months follow up, the only measurement that was found to not be statistically significant was the calcaneal pitch angle correction.

Analysis and Discussion

Triple arthrodesis incision planning has been described very seldom in literature.  In the study by Moore in 2014, a comparison was made between two groups of patients in which triple arthrodesis was attempted via one incision vs two [6]. The study showed that while there may not have been statistically significant differences between the two groups in regard to union rates; a similar result could be achieved in a more efficient amount of operating time.  They also proved that the TN joint could be accessed and prepared properly through a lateral incision.

In a study by Bono and Jacobs  evaluating triple arthrodesis performed through one lateral incision, the union rate of each joint was 80% in the STJ, 90% in the CC joint and merely 38% in the TN joint [7]. The authors of this study concluded that a triple arthrodesis could not be effectively achieved through one lateral incision.  

Despite the dependable and reproducible nature of arthrodesis, the disruption of soft tissue and ligamentous stability of the bones has been shown to cause complications.  These complications include wound dehiscence, delayed union, nonunion, re-operation and operating time. These complications as well as the benefits of decreased operating times were well described by Weinraub in their study in 2010 [4].  The study also took into consideration the addition of the fiscal benefits of decreasing cost by decreasing operating time. Similar complications were also found in our study as demonstrated by our complication rate. The comparisons of our study, while the focus is on radiographic measurements and patient satisfaction scores, help to build on these previous studies by producing repeatable and measurable results through the single incision.   

While joint preparation and visualization can prove to be more arduous from a lateral incision, this approach has been demonstrated in the past to be successful.  Limiting the amount of surgical incisions that need to heal can help to improve time of recovery in patients that may have poor soft tissue envelope. Despite the lack of some assessment on clinical outcomes, the radiographic data that was obtained by this study demonstrated that surgical correction can be made and maintained by using one lateral incision for triple arthrodesis.

The limitations of the case study were its retrospective and non-randomized nature.  There was also no control group for comparison. While the data collected centered around radiographic measurements and patient VAS scores as a representation of the outcome, there was a lack of clinical assessment of a valid outcome in the study.  The study was also limited by the lack of a long term follow up group for measurement and comparison of data. Potential considerations for future research include a comparison group of single incision vs. double incision patients of similar demographics.  

Patient Age Sex Diagnosis BMI Smoker PMH Complication
1 39 M PTTD 31.6 N HLD None
2 49 F PTTD 47 0.5 ppd CA Metal allergy

I&D, HWR

3 54 F Post-traumatic 35.4 N HLD, Asthma, Hypothyroid None
4 55 M PTTD 29.5 N None None
5 52 F PTTD 28.7 1 ppd Hypothyroid None
6 55 M Post-traumatic 21.8 N Depression None
7 23 M Spastic / Neurologic 19.6 N CVA None
8 68 F PTTD 41.5 N CA Non-union CC joint
9 68 F PTTD (Right) 37.6 N HTN, HLD None
10 68 F PTTD (Left) 37.6 N None None
11 16 M Midfoot Arthritis 24.3 N None None
12 53 F PTTD 42.1 N None None
13 43 F PTTD 48.7 N HTN, HLD HWR, dwyer osteotomy
14 56 F PTTD 32.4 N None None

Table 1 Patient Demographics  (N=14 operations in 13 patients). Abbreviations: BMI: Body mass index; PMH: Past medical history; PTTD: posterior tibial tendon dysfunction; ppd: Packs per day; HLD: hyperlipidemia; CA: Cancer; CVA: Cerebrovascular accident, I and D: Incision and Drainage; HWR: Hardware removal.

Average

Age

Gender Diagnosis BMI Smoker PMHx Complications
50 (16-68) 5 Males

(35.7%)

10 PTTD

71.4%)

34.1

(19.6-48.7)

2/14 (14%) as summarized above 3/14 (21.4%)
9 Females

(64.3%)

3 arthritis

(21.4%)

1 neurologic

(7.1%)

Table 2 Demographic summary.

Patient Pre-operative Immediate Post-operative Follow-Up
A B C A B C A B C
1 7 8 11 0 1 18 0 2 14
2 5 2 18 8 0 28 2 0 23
3 2 10 24 5 1 28 3 0 20
4 7 3 10 2 0 26 2 0 22
5 4 5 12 2 1 25 3 1 22
6 2 7 5 0 2 10 2 4 8
7 3 5 36 2 4 30 4 3 30
8 5 1 26 2 0 34 4 0 28
9 5 7 6 4 5 12 4 4 10
10 6 8 0 2 2 16 2 2 14
11 4 4 0 2 1 16 3 1 10
12 6 2 20 2 0 22 4 1 18
13 2 4 22 2 0 20 2 0 18
14 2 1 24 2 0 30 2 0 28

Table 3 Summary of Radiographic Measurements (Degrees). A: Talonavicular angle, B: Meary’s Angle, C: Calcaneal Pitch Angle.

Measurement  Immediate Post-op Six Months Post-op
TN Angle 0.02 0.0092
Meary’s Angle 0.0004 0.0004
Calcaneal Pitch Angle 0.0475 0.2978

Table 4 Summary of P-values Post Operatively.

References

  1. Myerson MS.  Triple Arthrodesis.  Reconstructive Foot and Ankle Surgery, Ed 2.  Edited by MS Myerson, Elsevier Saunders, Philadelphia, 2011.  
  2. McGlamry D, Ruch J, Mahan K, Napoli D.  Triple Arthrodesis. Podiatry Institute Update.  1987. Ch 30.
  3. Geocker RM, Ruch JA.  Triple arthrodesis. In McGlamry’s Comprehensive Textbook of Foot and Ankle Surgery, Ed 3.  Edited by AS Banks, MS Downey, DE Martin, SJ Miller, Lippincott Williams and Wilkins, Philadelphia, 2001.
  4. Weinraub GM, Schuberth JM, Leem, Rush S, Ford L, Neufield J, Yu J.  Isolated medial incisional approach to subtalar and talonavicular arthrodesis.  J Foot and Ankle Surg 2010; Vol. 49: 236-330.
  5. Jeng C, MD, Tankson C., Myerson, MD. The Single Medial Approach to Triple Arthrodesis: A Cadaveric Study.  Foot & Ankle Int 2006. Vol 27 (12).
  6. Blake E. Moore, MD, Nathaniel C. Wingert, MD, Kaan S. Irgit, MD,  Christian J. Gaffney, MD, and Gerard J. Cush, MD. Single-Incision Lateral Approach for Triple Arthrodesis.  Foot & Ankle Int 2014. Vol. 35: 896 –902.
  7. Bono JV, Jacobs RL. Triple arthrodesis through a single lateral approach: a cadaveric experiment. Foot Ankle. 1992. Vol. 13: 408-412.
  8. Berlet G, Hyer C, Scott R, Galli M.  Medial Double arthrodesis with lateral column sparing arthrodiastasis: A radiographic and medical record review.  J Foot and Ankle Surg 2015. Vol. 54: 441-444.
  9. Sammarco VJ, Magur EG, Sammarco GJ, Bagwe MR.  Arthrodesis of the subtalar and talonavicular joints for correction of symptomatic hindfoot malalignment.  Foot and Ankle Int. 2006. Vol. 27: 661-666.

Operating on patients with complex regional pain syndrome

by Ryon Wiska DPM1*, Lawrence Fallat DPM FACFAS2

The Foot and Ankle Online Journal 11 (2): 3

Complex regional pain syndrome (CRPS) is a debilitating disorder characterized by widespread, chronic pain. While elective procedures should be held until acute CRPS flare ups have subsided, certain scenarios require immediate surgical care. Surgical management of patients with CRPS requires a team approach with several other specialties including pain management and anesthesiology.  In this article, we outline a pre-operative and post-operative management course for lower extremity surgery of patients with diagnosed CRPS. We also present several case reports where this protocol was utilized.

Keywords: Causalgia, Complex Regional Pain Syndrome, CRPS, Pain, Reflex Sympathetic Dystrophy, RSD, Surgical Management

ISSN 1941-6806
doi: 10.3827/faoj.2018.1102.0003

1 – Second year podiatric surgery resident at Beaumont Hospital, Wayne.
2 – Program director for podiatric surgery at Beaumont Hospital, Wayne.
* – Corresponding author: rwiska@gmail.com


Physicians have been documenting disorders of chronic pain for centuries, with earliest documentation spanning back to Ambroise Pare’s description of chronic pain with King Charles IX in the 17th century [1]. Mitchell and colleagues documented cases of chronic pain in soldiers secondary to gunshot wounds and injuries of peripheral nerves during the Civil War [2]. Complex regional pain syndrome (CRPS) has historically been known by multiple names including reflex sympathetic dystrophy, causalgia, Sudeck’s atrophy, and shoulder-hand syndrome. Most experts now abide by terminology introduced by the International Association for Study of Pain (IASP) in 1994, which subdivided CRPS into type 1 and type 2, with type 2 having an inciting nerve injury [3].

The diagnosis of CRPS is based on clinical findings. The original IASP diagnostic criteria for CRPS includes: 1) The presence of an initiating noxious event or a cause of immobilization. 2) Continuing pain, allodynia, or hyperalgesia with which the pain is disproportionate to any inciting event. 3) Evidence at some time of edema, changes in skin blood flow, or abnormal sudomotor activity in the region of pain. 4) This diagnosis is excluded by the existence of conditions that would otherwise account for the degree of pain and dysfunction [4]. More recent literature from the Reflex Sympathetic Dystrophy Association unveiled a clinical diagnostic criteria update, which reflects systemic findings that can be documented during patient visits (Table 1)[5]. Current management for active CRPS includes physical therapy, antidepressant agents, gabapentin, corticosteroids, topical analgesics, opioids, sympathetic blocks, somatic blocks, and neuromodulation [6-10].

  1. Continuing pain, which is disproportionate to any inciting event
  2. Must report at least one symptom in three of the four following categories Sensory: Reports of hyperalgesia and/or allodynia
    1. Vasomotor: Reports of temperature asymmetry and/or skin color changes and/or skin color asymmetry
    2. Sudomotor/Edema: Reports of edema and/or sweating changes and/or sweating asymmetry
    3. Motor/Trophic: Reports of decreased range of motion and/or motor dysfunction (weakness, tremor, dystonia) and/or trophic changes (hair, nail, skin)
  3. Must display at least one sign* at time of evaluation in two or more of the following categories
    1. Sensory: Evidence of hyperalgesia (to pinprick) and/or allodynia (to light touch and/or deep somatic pressure and/or joint movement)
    2. Vasomotor: Evidence of temperature asymmetry and/or skin color changes and/or asymmetry
    3. Sudomotor/Edema: Evidence of edema and/or sweating changes and/or sweating asymmetry Motor/Trophic: Evidence of decreased range of motion and/or motor dysfunction (weakness, tremor, dystonia) and/or trophic changes (hair, nail, skin)
  4. There is no other diagnosis that better explains the signs and symptoms

* A sign is counted only if it is observed at time of diagnosis

Table 1 Clinical diagnostic criteria for complex regional pain syndrome.

In recent pain management literature, low dose naltrexone (LDN) has been shown to be efficacious in treating patients with CRPS [11].  LDN refers to doses approximately 50-fold lower than doses of naltrexone typically given to patients addicted to opioids [12]. It has been shown that LDN antagonizes the Toll-like receptor 4 (TLR 4) pathway and attenuate microglia. TLR 4 in both CNS neurons and microglia augments the production of pro-inflammatory cytokines via the nuclear factor kappa-light-chain-enhancer of activated B cells (NF- κB) pathway, which acts as a mediator for neuropathic pain [13]. In a double blind study of 30 women with chronic pain, twice daily administrations of 4.5 mg of Naltrexone resulted in 57% of the participants exhibiting a significant reduction in pain when compared to placebo [14].

The current paradigm of surgeons has been to avoid operating on patients with CRPS because symptoms could either recur or worsen. In 6-10% of patients, surgical intervention is warranted and should not be delayed. These conditions include painful deformity, displacement of fixation, fracture, trauma, tumors, and soft tissue masses [15].  Surgical management of patients with CRPS has been documented in orthopedic literature, with several papers discussing surgical intervention of the upper extremity and the knee [16,17]. Previous recommendations in the knee included waiting 5 months, with ranges from 2 months to 1 year. Prolonging surgery allows for subsidence of acute pain, as well as allowing time for treatment such as sympathetic blocks and physical therapy [16,17]. The purpose of this article is to outline a surgical management approach preoperatively and postoperatively for patients with active CRPS to provide the podiatric surgeon with management options, as well as review three cases in which this protocol was used.

Preoperative Management

Surgical management of patients with CRPS requires a team approach. It is imperative to coordinate with the physician who is actively managing the patient’s CRPS. If a patient does not have an active pain management specialist, consultation with a pain management specialist should be sought prior to operating. The surgeon should coordinate with the pain management physician as well as anesthesiologist regarding patient’s operative management course with clear understanding of preoperative and postoperative treatment.

Recent literature has found that low dose naltrexone (LDN) has been shown to be efficacious in treating patients with CRPS through its endorphin releasing and anti-inflammatory properties. However, it should be held at least 24-36 hours before surgery to ensure that opiate medication administered from anesthesia is able to reach full efficacy.

Anesthesia choice is critical in ensuring that CRPS flare-ups or increase in symptoms of active CRPS do not occur. Either epidural or spinal anesthesia should be utilized in patients with CRPS. This practice has been documented in orthopedic literature with studies showing recurrence rates of CRPS falling from 72% to 10% with the use of a preoperative spinal or epidural block [16]. It is believed that this may provide a clinical advantage by blocking the potential barrage of nociceptive afferent signals in the central nervous system during surgery [18].

When trying to evaluate whether epidural or spinal anesthesia should be performed, several variables should be considered such as time from start of induction to achievement of anesthesia, time for resolution of anesthesia, and possible side effects. In spinal anesthesia, the average time from intrathecal injection of local anesthetic to achievement of surgical anesthesia is 13 minutes in conventional spinal and 16 minutes in unilateral spinal [19]. In epidural anesthesia with insertion of catheter, the average time to achieve induction was noted to be 40 minutes [20]. When performing spinal anesthesia with 5 mg hypobaric bupivacaine, Ben-David reported two segment regression after 53 minutes and discharge after 180-190 minutes, with newer studies stating average PACU time following completion of procedure ranging between 65-98 minutes [21, 22]. Mulroy noted average PACU time for patients who received epidural prior to knee surgery as 92 ±18 minutes [23].

Epidural allows for the titration of short term local anesthetic which may lead to quicker discharge times following outpatient procedure, while still providing blockade to prevent CRPS flare-ups. In patients with CRPS, epidural catheter allows the option for continuous titration of anesthetic, which may be beneficial following the procedure, whereas spinal anesthesia employs a single dosage of anesthetic.

Common side effects noted in both spinal and epidural anesthesia are hypotension, bradycardia, post-dural puncture headaches, nausea, and vomiting.  In more severe side effects, prolonged neurological complications have been observed. In epidural anesthesia, urinary retention is also a common side effect, which may require catheterization and hospitalization.

Postoperative Management

Following the surgical procedure, patients are admitted for 24-48 hours of IV pain medication administration. Patients are given take-home oral analgesic medication for pain relief until acute surgical pain has subsided. Typical examples of oral medications include Percocet 10 mg/325 mg, 1-2 tablets by mouth every 4-6 hours, or Hydromorphone 2-4 mg, 1 tablet by mouth every 4-6 hours. If patients were previously taking LDN, they are to resume daily LDN when surgical pain is controlled and after 7 days have elapsed. We recommend early range of motion and aggressive physical therapy following procedure once the surgical site is stable. If symptoms of CRPS appear to be exacerbated following surgery, we recommend patients undergo intravenous Ketamine infusion therapy, under the management of their pain specialist.

Case Report No. 1

A 38-year-old male presented to our clinic one month after injuring his right foot when a 1000-pound roll of vinyl fell onto his foot. The patient was initially referred to our clinic for care of a nondisplaced fracture of the fifth metatarsal; however, radiographs and bone scan failed to reveal signs of fracture and a diagnosis of contusion to the right foot was made. The patient had been immobilized in a nonweightbearing below knee cast for one month and had subsequently developed increased pain out of proportion to injury as well as exhibited mottling of skin to dorsum of right foot in relation to left. The patient also began to exhibit rigid contractures of the right tibialis anterior, extensor digitorum longus, and extensor hallucis longus.  The patient was referred to a pain management specialist where the diagnosis of CRPS to the right lower extremity was made. The patient reported that since date of injury, the pain had progressively increased and at time of initial presentation, was so severe that even light touch to the right lower extremity was excruciatingly painful. On evaluation it was determined that the patient exhibited two distinct types of pain, a generalized CRPS pain to the affected lower extremity as well as a muscular pain secondary to rigid contractures. The patient was treated in our office monthly for peripheral nerve blocks at the level of the ankle joint consisting of 0.5% Marcaine plain, which the patient reported provided several hours of relief of contractures and pain before pain and contractures returned.  At the same time, he underwent 22 sympathetic blockades over the course of 3 years from 4 different pain clinics, but had no relief, despite multiple pain management treatment modality attempts including a spinal cord stimulator. The patient was treated noninvasively by pain management specialists as well as our clinic for approximately 3 years, at which time it was determined that pain level had plateaued and was not improving with the treatment.

The patient underwent three different manipulations of the right foot under epidural anesthesia. The extensor tendons were stretched for a period of 20 minutes, until relaxation of rigidly contracted muscles were noted. The patient was then placed in an anterior splint following the procedures. No acute flare up of CRPS was seen immediately after the procedure; however, the patient exhibited return of rigid contractures and pain 48 hours following each procedure and was unable to tolerate the anterior splint. No increase in CRPS pain was seen following procedures.

The patient then underwent a series of botox injections that provided some pain relief and reduction of contracture to right foot and ankle that lasted for approximately two to three weeks before the muscles returned to rigid clonus.

Surgical intervention to the right foot was discussed with the patient. The patient was offered procedures that included manipulation of the right foot under anesthesia, capsulotomy of the right first metatarsophalangeal joint, lengthening of the tibialis anterior, extensor hallucis longus, and extensor digitorum longus tendons, and sectioning of extensor hallucis brevis, all of the right foot. The patient was advised that procedure may exacerbate symptoms of CRPS and that no guarantees were given or implied. The patient met with his pain management specialist prior to the procedure and was given provisions for oral analgesics following the procedure.

On the day of the procedure, anesthesia was obtained with spinal anesthesia. The patient was placed on the operating table in the supine position. Manipulation of the foot was first performed where attention was made to manually plantarflex the right ankle joint as well as toes 1-5, which were noted to be rigidly contracted in a dorsiflexed position. Following manipulation, the foot was noted to held in a plantarflexed position. Standard z-lengthening procedures were then performed to the extensor digitorum longus, extensor hallucis longus, and tibialis anterior tendons. The extensor hallucis brevis tendon was identified and then sectioned proximal to its insertion.  Attention was then directed to the first metatarsophalangeal joint where a dorsal and lateral capsulotomy was then performed and contracture of the first metatarsophalangeal joint was noted to be decreased. Closure was completed using a combination of dissolving and nondissolving suture. A postoperative block was then infiltrated around the incision sites consisting of 9 mL of 0.5% Marcaine plain and 2 mL of dexamethasone.

Following the procedure, the patient was admitted for 48-hour pain management. The patient reported a relief in pain following the procedure and was able to tolerate weight bearing to the right lower extremity without the use of an assistive device for the first time since the injury. Ultimately, the patient reported return of CRPS pain and contractures 2 weeks following the procedure; however, no increase in CRPS pain was noted. In addition, the patient noted that contractures to the right lower extremity were not as rigid or painful.

Case Report No. 2

A 31-year-old female with history of CRPS type 1 after sustaining multiple injuries from a motor vehicle accident presented to our clinic with complaints of right ankle pain. The patient had history of multiple surgeries to her right ankle with internal fixation after suffering a comminuted open right ankle fracture. The patient’s pain was actively cared for by a pain management specialist who had maintained the patients pain in a tolerable level with the use of LDN as well as IV Ketamine infusion therapy. The patient presented to our office with complaints of a painful right ankle, which had subsequently developed a severe valgus alignment of the right heel, subtalar joint arthritis, a nonunion of a right fibular fracture, as well as pain along course of retained hardware. Despite active pain management therapy, the patient admitted to 10/10 pain to the right ankle.  The patient related that pain to her right ankle was becoming debilitating to the point that she was unable to ambulate. Initial attempts were made to treat the patient conservatively with the use of padding, bracing, and offloading with patient reporting no relief of pain. When conservative treatment options were exhausted, the patient was advised of surgical correction. The patient was made aware that surgical correction risked the possibility of a CRPS flare-up. She was fully aware of this and wished to proceed with procedure. Prior to boarding procedure, multiple conversations were had with patient’s pain management specialist as well as anesthesia team at our institution with preoperative, perioperative, and postoperative management discussed at length. It was determined that prior to procedure, patient was to hold LDN. The day of the procedure, patient was to obtain a popliteal block prior to induction and then undergo general anesthesia. The patient was then to be admitted for extended stay pain monitoring.

Twenty-four hours prior to procedure, patient’s LDN was held. On the day of the procedure, the patient was to undergo a popliteal block prior to induction; however, she did not receive the block prior to procedure. The patient was brought to the operating room and placed on the hospital table in the supine position where general anesthesia was obtained. The patient then underwent removal of painful retained bone screws and plates of the ankle, open reduction and internal fixation of right fibular nonunion, resection of synostosis of right ankle, excision of scar tissue of right ankle, medial transpositional calcaneal osteotomy with internal fixation of right foot, as well as arthrotomy of right ankle.  The patient was then placed in a well-padded cast and was instructed to be non-weight bearing to the right lower extremity with the use of crutches.

Following the procedure, the patient awoke from anesthesia in intense pain to the surgical limb. An epidural was placed and pain was controlled. The patient was converted to 48 hour full admit due to epidural. After 24 hours, she related that epidural was starting to wear off and was admitting to increased pain to surgical limb. The patient was maintained on IV Dilaudid and oral Percocet, 10 mg. After 4 days postoperatively, her pain was maintained on oral Percocet and patient was discharged home.  The patient went on to achieve surgical union of fibular fracture, but continued to admit to CRPS pain to the surgical limb, which limited activities of daily living. The patient related to no increase in CRPS pain. Six months following her procedure, the patient successfully underwent a spinal cord stimulator trial. Following insertion of the stimulator, the patient was able to stand and walk around a department store, which she had been unable to do following the initial accident. Although the patient still relates to CRPS pain, the pain related to her foot and ankle condition has subsided and no increase in CRPS pain has been noted.

Case Report No. 3

A 65-year-old female presented to our office with history of CRPS, which she developed following a third intermetatarsal space neurectomy to the left foot. On clinical exam, the patient exhibited symptoms of a neuroma to the second intermetatarsal space to the left foot as well as a stump neuroma to the third intermetatarsal space of the left foot and admitted to 10/10 left pain with maximal tenderness to the forefoot. The patient admitted that pain to the left foot was so intense that her ability to ambulate was becoming limited. Conservative treatment was attempted with offloading, padding, and local steroid injections to the affected intermetatarsal spaces, which provided little to no relief. Once conservative options had failed, surgical intervention was discussed with the patient.  The patient was advised that the proposed procedures would be an excision of neuroma to second and third intermetatarsal space of the left foot. The patient was made aware that CRPS symptoms could be exacerbated by the procedure and that clear pain management goals were outlined with her pain management physician.

On the day of the surgery, anesthesia was obtained with spinal anesthesia as well as a local anesthetic block to the second and third intermetatarsal spaces of the left foot. Anatomic dissection was carried down to the level of the neuroma and nerve was tracked proximally until healthy nerve tissue was observed. Inflamed nerve was then resected from the second and third intermetatarsal space. A 4 cm x 2 cm x 0.5 cm nerve specimen was excised from the second intermetatarsal space and a 2 cm x 1.5 cm x 0.3 cm nerve specimen was excised from the third interspace.  Closure was then performed with a combination of dissolving and non-dissolving suture and a postoperative block was infiltrated to the incision site consisting of 9 mL of 0.5% Marcaine plain and 4 mL of dexamethasone. The patient was given Norco 7.5 mg/325 mg for pain control postoperatively and was partial weight bearing to the left heel in a surgical shoe. The patient declined postoperative observation for pain management and was discharged home once cleared by anesthesia.

Following the procedure, the patient reported no increased exacerbation of CRPS and admitted to decreased pain to the neuroma site on the left foot. While the patient still reports CRPS pain to the left lower extremity, she is now able to pursue activities of daily living and maintains a tolerable level of pain to the left lower extremity.

In conclusion, our outlined pre-operative and post-operative management course for lower extremity surgery of patients with diagnosed CRPS has proven effective in preventing flare-ups of CRPS and preventing increase of active CRPS pain.

Funding Declarations

No funding was used.

Conflict of Interest

None

References

  1. Pare A. Of the cure of wounds of the nervous parts. In The Collected Works of Ambroise Pare, book 10, pp 400-402, translated by T Johnson, Milford House, Pound Ridge, NY, 1634.
  2. Mitchell SW; Morehouse GR,  Kenn WW. Gunshot Wounds and Other Injuries of Nerves, pp 148-157, JB Lippincott, Philadelphia, 1864.
  3. Stanton-Hicks M, Baron R, Boss R, et al. Complex regional pain syndrome: guidelines for therapy. Clin J Pain. 1998; 14:155-166.
  4. Merskey H, Bogduk N. Classification of Chronic Pain: Descriptions of Chronic Pain Syndromes and Definitions of Pain Terms, 2nd edition. Seattle, WA: IASP Press; 1994.
  5. Harden, R. N., Oaklander, A. L., Burton, A. W., Perez, R. S. G. M., Richardson, K., Swan, M., Barthel, J., Costa, B., Graciosa, J. R. and Bruehl, S. (2013), Complex Regional Pain Syndrome: Practical Diagnostic and Treatment Guidelines, 4th Edition. Pain Med, 14: 180–229. doi:10.1111/pme.12033
  6. Rho RH, Brewer RP, Lamar TJ, Wilson PR. Complex regional pain syndrome. Mayo Found Med Educ Res 77: 174-180, 2002.
  7. Oerlemans HM, Oostendorp RA, de Boo T, van der Laan L, Severens JL, Goris JA. Adjuvant physical therapy versus occupational therapy in patients with reflex sympathetic dystrophy/complex regional pain syndrome type I. Arch Phys Med Rehabil. 2000; 81:49-56.
  8. Kingery WS. A critical review of neuropathic pain: antidepressants and opioids. Clin J Pain. 2000; 16 (2, suppl):S49-S55.
  9. Kemler MA, Barends GAM, van Kleef M, et al. Spinal cord stimulation in patients with chronic reflex sympathetic dystrophy. N Engl J Med. 2000; 343:618-624.
  10. Van Hilten BJ, van de Beek W-JT, Hoff JI, Voormolen JHC, Delhaas EM. Intrathecal baclofen for the treatment of dystonia in patients with reflex sympathetic dystrophy. N Engl J Med. 2000; 343: 625-630.
  11. Chopra P, Cooper M. Treatment of complex regional pain syndrome using low dose naltrexone. J neuroimmune pharmacol. 2013; 8:470-476.
  12. Rea F, Bell JR, Young MR, Mattick RP. A randomized controlled trial of low dose naltrexone for the treatment of opioid dependence. Drug Alcohol Depend. 2004; 75:79-88.
  13. Milligan ED, Watkins LR. Pathological and protective roles of glia in chronic pain. Nat Rev Neurosci. 2009; 10:23-26.
  14. Younger J, Noor N, McCue R, Mackey S (2013) Low-dose naltrexone for the treatment of fibromyalgia: findings of a small, randomized, double-blind, placebo-controlled, counterbalanced, crossover trial assessing daily pain levels. Arthritis Rheum, 2013; 65(2):529–538
  15. Moesker A: Complex regional pain syndrome, formerly called Reflex sympathetic dystrophy, treated with Ketanserin and Carnitine (thesis). Rotterdam, Erasmus University Rotterdam, 2000, pp 1-147.
  16. Reuben S, Rosenthal E, Steinberg R. Surgery of the affected upper extremity of patients with a history of complex regional pain syndrome: a retrospective study of 100 patients. J of Hand Surgery. 2000; 6:147-151.
  17. Katz MM, Hungerford DS:  Reflex Sympathetic Dystrophy Affecting the knee.  J Bone Joint Surg. 1987; 69: 797 – 803.
  18. R. Norman Harden, MD, Stephen Bruehl, PhD, Michael Stanton-Hicks, MB, BS, DMSc, FRCA, ABPM, Peter R. Wilson, MB, BS; Proposed New Diagnostic Criteria for Complex Regional Pain Syndrome. Pain Med 2007; 8 (4): 326-331.
  19. Fanelli, G, Borghi, B, Casati A, et al. Unilateral bupivacaine spinal anesthesia for outpatient knee arthroscopy. Can J Anesth 2000; 47 (8):746-751.
  20. Koenig T, Neumann C, Ocker T, Kramer S, et al. Estimating the time needed for induction of anesthesia and its importance in balancing anaesthetists’ and surgeons’ waiting times around the start of surgery. Anaesthesia 2011; 66:556-562.
  21. Ben-David B, Levin H, Solomon E, Admoni H, Vaida S. Spinal bupivacaine in ambulatory surgery: the effect of saline dilution. Anesth Analg 1996; 83: 716–20.
  22. Valanne J, Korhonen AM, Jokela R, Ravaska P. Selective spinal anesthesia: A comparison of hyperbaric bupivacaine 4 mg versus 6 mg for outpatient knee arthroscopy. Anesth Analg 2001; 93: 1377-9.
  23. Mulroy M, Larkin K, Hodgson P, et al. A comparison of spinal, epidural, and general anesthesia for outpatient knee arthroscopy. Anesth Analg 2000; 91:860-4.

The vacuum phenomenon in the ankle joint: Air bubbles on CT

by Christopher R. Hood JR. DPM1*, Wesley A. Jackson DPM2, Robert C. Floros DPM3, David A. Bernstein, DPM4

The Foot and Ankle Online Journal 11 (2): 2

Gas or air bubbles in a joint space are most commonly associated with the “vacuum phenomenon,” a collection of gas that has precipitated out of solution to take up a gaseous state within a joint. This phenomenon was unbeknown to us upon a patient presentation, seen on computed tomography scan, and so further academic investigation was performed to define this pathology. Because of this lack of awareness, a PubMed® literature review was performed to analyze the rate of incidence in foot and ankle. Additionally, we present a case example of the vacuum phenomenon in the ankle joint of a 50-year old patient presenting with degenerative ankle joint pain symptoms. Further, a review of the condition as well as differentials is discussed in an attempt to raise awareness of this differential diagnosis for gas bubbles within a joint.

Keywords: Air bubbles, ankle, arthritis, CT, computed tomography, foot, gas bubbles, gaseous degeneration, vacuum phenomenon

ISSN 1941-6806
doi: 10.3827/faoj.2018.1102.0002

1 – Fellowship-Trained Foot and Ankle Surgeon, Premier Orthopaedics and Sports Medicine, Malvern PA.
2 – Resident, PGY-2, Bryn Mawr Hospital PMSR/RRA, Bryn Mawr PA.
3 – Private practice, Ocean County Foot & Ankle Surgical Associates, P.C., Toms River NJ and The Foot and Ankle Center, Haverford PA.
4 – Residency Director, Bryn Mawr Hospital PMSR/RRA, Bryn Mawr PA and Private Practice, Wayne PA.
* – Corresponding author: crhoodjr12@gmail.com


The presence of gas or air bubbles in a joint was first described by Fick in 1910 when he noticed gas bubbles in hand joints on radiograph (XR) evaluation while under traction [1-3]. Later this radiographic finding was coined the “vacuum phenomenon” (VP) by Magnusson in 1937 [2]. On imaging modalities such as computed tomography (CT) where it is most often visualized, it appears as a dark radiolucent pattern that can be shaped anywhere between a singular, linear bubble to confluence of bubbles within the confines of a joint space [2]. The shape is classically defined as a crescentic lucency paralleling a joint when found articular [3].

Gas bubbles were first thought to be associated with joint traction or trauma, but has since been found in situations of degenerative changes to joints [1,2]. Other associated gas bubble presenting pathologies include fracture-dislocation (e.g. traction injuries, open injuries introducing free air), ligament injury, metastasis, infection (e.g., abscess, osteomyelitis), cancer (e.g., multiple myeloma), intervertebral disc herniation/Schmorl’s nodes, abdominal or thoracic free air (e.g., digestive tract perforation, pneumothorax, air embolism), decompression sickness, and iatrogenic causes (e.g., surgical introduction of air, arthroscopy) [2,4].

Related to degenerative disease, its presence is most often cited to the sacroiliac (SI) joints (i.e., joint, facets, intervertebral discs) but also has been found in the pubic symphysis, lumbosacral space, and the joints of the temporomandibular, wrist, hand, hip, shoulder, knee, ankle (AKJ), subtalar (STJ), and calcaneocuboid (CCJ) [1-4]. Analysis of the gas in the SI location has found it to be predominantly nitrogen (> 90%) based , but oxygen and carbon dioxide among other gases are also present at much lower concentrations [2].

The purpose of this report was twofold: to determine the rate of occurrence of foot and/or ankle VP in the literature through a keyword search and present a case example of the VP to the AKJ in an end-stage degeneration clinical situation.

Methods

A PubMed® advanced keyword search was performed on May 1, 2017,  using the term combinations of “air bubble,” “bubble,” “gaseous degeneration,” “vacuum phenomenon,” with “foot” or “ankle.” The search had no restriction parameter fields applied. (Table 1) The returned abstracts were reviewed to determine their validity whether relevant to the primary search goal of obtaining articles demonstrating the VP from the ankle joint, distally. A table was then created counting the published instances of the VP in the foot and/or ankle.

Case Report

A 50-year old male patient presents to the senior author’s office after referral from a previous podiatrist due to his primary complaint of ankle pain. The patient described the pain as a progressive pain upon ambulation. The patient is very active and enjoys running and mountain climbing in particular. He states he can walk up to 8 miles until he can’t bare the pain anymore. He states his pain has been progressing in the ankle for 8 years now. Only rest has been able to alleviate his symptoms to this point in time. He has not sought any formal medical treatment prior to presentation.

Figure 1 50 year-old male, sagittal CT scan of the ankle. Note the gas formation in the joint as well as presence within the subchondral bone region. Associated talar dome arthritic changes. Images are left to right, lateral to medial.

Figure 2 50 year-old male, coronal CT scan of the ankle. Note the gas formation centered around, and within the cystic changes to the medial talar dome. Images are left to right, anterior to posterior.

Figure 3 50-year-old male, axial CT scan of the ankle. Note the gas formation is positioned with the lower-lying cartilage defect space. Images are left to right, superior to inferior slices.

Ankle Foot
  • “air bubble” / “ankle”
  • “air bubble” / “foot”
  • “bubble” / “ankle”
  • “bubble” / “foot”
  • “gaseous degeneration” / “ankle”
  • “gaseous degeneration” / “foot”
  • “vacuum phenomenon” / “ankle”
  • “vacuum phenomenon” / “foot”

Table 1 Key Word Search Parameters for Study Identification – Vacuum Phenomenon to the Foot and/or Ankle.

Ankle Foot
  • “air bubble” / “ankle” = 0
  • “air bubble” / “foot” = 1
  • None related to topic
  • “bubble” / “ankle” = 3
  • None related to topic
  • “bubble” / “foot” = 21
    • 1 discussing foot drop  developed 10 days post-op disc surgery, secondary to nerve root gas bubble (Kloc et al., 1998)(6)
    • 1 discussing STJ ROM using a bubble inclinometer
    • 1 discussing diabetic foot bullosis diabeticorum
  • “gaseous degeneration” / “ankle” = 0
  • “gaseous degeneration” / “foot” = 2
    • 1 discussing foot drop  developed 10 days post-op disc surgery, secondary to nerve root gas bubble (Kloc et al., 1998)(6)
  • “vacuum phenomenon” / “ankle” = 4
    • 1 related to dislocated joint/trauma of STJ and CCJ (Ahmad et al., 2007)(5)
    • 1 related to STJ and AKJ (Lee et al., 1994)(1)
    • 2 discussing lumbar pathology
  • “vacuum phenomenon” / “foot” = 2
    • 1 related to STJ and AKJ (Lee et al., 1994)(1)
    • 1 discussing foot drop  developed 10 days post-op disc surgery, secondary to nerve root gas bubble (Kloc et al., 1998)(6)

Table 2 Study Search Resulted Literature – Vacuum Phenomenon to the Foot and/or Ankle.

The patient’s past medical history consists of hemochromatosis. There is no known past surgical history to the foot or ankle. There is no known family history of foot or ankle pathologies at this time. Medications consist of hydrochlorothiazide and a baby aspirin daily.

The patient’s physical exam findings show limited dorsiflexion at the ankle joint and pain upon end range of motion in dorsiflexion at the ankle joint with a hard stop. His neurovascular status was grossly intact. There were no subjective complaints or objective findings of an infectious process based on the history and physical exam. He had no complaints of any other arthritic or painful joints. No other abnormalities were noted to his problem based exam.

A CT scan of the ankle exhibited degenerative joint disease to the talotibial joint along with a large anterior osteophyte of the distal tibia and talar neck at the ankle joint level. The CT scan also exhibited intra-articular gas centrally within the joint (Figures 1-3). Upon discussion with the reading radiologist it was declared that the gas was related to the VP. Further discussion with multiple facility radiologists where the study was performed revealed that the gas is due to nitrous oxide from surrounding synovial tissues, but can also be due to positioning of the ankle joint at the time of the study. From their experience, most VPs noted by these radiologists occur primarily in the lumbar spine and shoulders. None of them have seen such a finding in the ankle until this particular case.

Discussion of treatment options with the patient included less impacting exercises, an anterior ankle joint arthroplasty, and the need for a possible ankle joint replacement in the long term future. The patient was in favor of the anterior ankle arthroplasty procedure but would take time to think about his options moving forward. No treatments have been rendered to date and he has not returned to the senior authors’ facility.

Results

From the PubMed® literature search, 33 articles resulted in total. After reviewing titles, abstracts, and database tags, removing irrelevant and duplicate entries, only two articles were relevant to this literature review of identifying examples of the VP in the foot and/or ankle (Table 2). This included a retrospective institutional review of CT imaging over two years evaluating the presence of gas bubbles in the lower extremity joints (i.e., AKJ, STJ, CCJ) and a case example of the VP in the STJ and CCJ after a trauma [1,5]. A third study found discussed a drop foot secondary to epidural gas formation and nerve root compression was not counted due to the distance location of the gas bubbles from the foot [6].

Discussion

The VP is a combination of anatomy and physics, calling into play both Henry’s Law and Boyle’s Law through hydrodynamic cavitation [2,4]. Simply put, gas precipitates out of solution through a negative intra-articular pressure when a joint is distended (e.g. traction) or collapses. The newly created free space within the joint capsule needs to be filled, and is done so by gas (primarily nitrogen) [2,3]. In this situation it is often by a gaseous element that precipitates out of the local tissue or synovial fluid due to changes in pressure [1,2,4]. Gohil et al (2014) and Yanagawa et al (2016) provide detailed explanations of this phenomenon. Normally, this gas goes back into solution when the joint returns to its normal volume and pressure. However in situations of arthritis, a thickened or fibrotic/scarred joint capsule does not allow the gas to dissolve out. Furthermore, excess joint space due to the presence of cartilage loss and subchondral cysts allows the gas to remain out of solution to fill that “extra” space [1]. In situations of traction or trauma to a joint,  the blood gas nitrogen precipitates out of solution to fill the excess free intra-articular space from the joints’ distention [5]. In open fractures, the outside air fills the spaces within the extremity, and is not a true VP.

The presence of the VP may be seen as something no more than an academic finding when present on a CT scan of a lower extremity joint. It has been documented in instances related to trauma (i.e. sprain; joint dislocation; rapid joint distention,) degenerative disease, osteochondrosis, osteonecrosis, idiopathic, osteomyelitis / infection, or conditions specific to the joint found in [1,2,5]. Its finding is most often related to degenerative disease to a joint, easily seen on CT due to its greater sensitivity  with higher resolution compared to XR or magnetic resonance imaging (MRI) [1,2,4]. Associated pathology such as narrowed space, subchondral cyst, sclerosis, hypertropic degeneration to the joint may be seen along with the gas bubbles in degenerative situations across each imaging modality. In acute trauma, the presence of gas would suggest intact joint capsule and the associated intra-capsular ligaments however reports in the knee have shown otherwise [2].

When found, one important point is to correlate the finding to the presenting pathology through the patient history and physical exam so to not over or under diagnose the true pathology at hand [2,3]. This is most important when wanting to rule out any potential infectious processes such as septic joint, open fracture-dislocation, or penetrating joint trauma. Joint gas and spinal infection has been associated with bacteria such as obligate anaerobes or facultative organisms such as clostridia, Peptococcus, and E. coli [1,7]. Patterns of gas formation have been cited with different pathology from a linear formation in more benign pathology while bubble-like multi-lobulated patterns suggest infection [2]. In closed injuries, the presence may suggest a recent joint dislocation that otherwise may not be visible on imaging [5].

Specific to the lower extremity, Lee et al. (1994) performed an institutional retrospective review of CT scans over a two year period  to determine the incidence of gas within the STJ and/or AKJ [1]. It was documented in 12 cases (n = 495, 2.4%) on CT, none of which were related to infection. Of these, 11 were in situations of arthritis (post-traumatic, 10; non-traumatic, 1), 10 cases in the STJ, and although the XR did not show gas or air in the joints, degenerative changes were present and visible on both XR and CT. In the only other example, Ahmad et al. (2007) demonstrated the VP in a single case of an acute, closed STJ and CCJ fracture-dislocation [5]. One final unrelated  but interesting case included epidural gas collection secondary to vertebral disc degeneration causing nerve root compression and a drop foot [6]. Ultimately, surgical decompression resulted in resolution of the drop foot.

The VP is very under-reported in the literature and in radiology reports [2]. In the SI joint where the finding is most common, one study found only a 16% reported rate [2]. For the case presented here, the finding was not mentioned in the radiologists report. Only in calling the radiologist who performed the evaluation did we get an explanation of the gas finding seen on CT. The condition may be unfamiliar to physicians other than radiologist, as was in this instance, where more awareness would be important for the ordering physician to add the VP to their differential diagnosis of gas in a joint without jumping directly to infection [2,7].

The authors surmised the VP finding in the lower extremity may not be seen in high percentages due to two more reasons. These are based on the physics of the VP and some speculation [2]. The first is that the VP is most sensitive on CT imaging. In instances of acute trauma to the lower extremity such as traction injuries (i.e. sprains) that are often evaluated, diagnoses, and treated in the outpatient setting, an MRI is often the modality used if advanced imaging is required. In these traumatized joints, by the time imaging is performed, the gas has possibly gone back into solution and fluid fills any remaining excess intra-articular space. In acute injury settings such as joint dislocations, it has been suggests that gas bubbles may be routinely seen within 4 hours of dislocation while occasionally seen after 48 hours on CT scan [8]. Another multi-joint study found, after inducing a transient traction-VP, the gas bubbles to disappear within 10 minutes [3]. If acute fracture-dislocations present in the emergent setting and the more sensitive CT is ordered, the VP finding may often be overlooked due to the more pressing osseous trauma that requires urgent treatment or be attributed to a concomitant open injury and free air. Second relates to the duration of gas presence in a joint, other than the aforementioned points. In situations of chronic degenerative disease, over time the gas within the joint achieves a new solubility equilibrium and will dissolve back into solution and not be visible. The time to reach equilibrium was not found in any report.

Conclusion

The VP is a finding consisting of gas or air bubbles on CT within a joint space. Its finding is under represented in the lower extremity joints with only two citations to date (not including this report). The presence should not be alarming when seen in a non-infectious presentation. Although its finding to date is not correlated with a more advanced joint degeneration to the lower extremity, the finding can be another example of degeneration in addition to visible cartilage loss, subchondral cysts, and scarred joint capsules. This example adds to the literature base of VP to the lower extremity and provides another mode of bringing awareness to physicians who treat the lower extremity.

Financial Disclosures / Funding Declaration

None

Conflict of Interest

None

Acknowledgements

None

References

  1. Lee TH, Wapner KL, Mayer DP, Hecht PJ, D M. Computed tomographic demonstration of the vacuum phenomenon in the subtalar and tibiotalar joints. Foot Ankle Int. 1994;15(7):382–5.
  2. Gohil I, Vilensky JA, Weber EC. Vacuum phenomenon: clinical relevance. Clin Anat. 2014;27:455–62.
  3. Balkissoon ARA. Radiologic interpretation of vacuum phenomena. Crit Rev Diagn Imaging. 1996;37(5):435–60.
  4. Yanagawa Y, Ohsaka H, Jitsuiki K, Yoshizawa T. Vacuum phenomenon. Emerg Radiol [Internet]. Emergency Radiology; 2016;23:377–82. Available from: http://dx.doi.org/10.1007/s10140-016-1401-6
  5. Ahmad R, Annamalai S, Radford M, Cook C. Vacuum phenomenon in a dislocated joint. Emerg Med J. 2007;24:862.
  6. Kloc W, Wasilewski W, Imieliński B, Karwacki Z. Epidural gas aggregation in the course of gaseous degeneration of lumbar intervertebral disk as a cause of foot paresis. Neurol Neurochir Pol. 1998;32(3):699–704.
  7. Nagashima T, Minota S. Air bubbles in the knee joint. J Clin Rheumatol. 2016;22(2):94–5.
  8. Fairbairn KJ, Mulligan ME, Indication A, Fairbairn KJ, Murphey MD, Resnik S. Gas bubbles in the hip joint on ct: an indication of recent dislocation. Am J Roentgenol. 1995;164(May):931–4.

Progression of a digital or partial ray amputation to transmetatarsal amputation and below knee amputation: Time frames and associated comorbidities, a three-year retrospective study

by Carmen Bruno DPM1*, Susan Wiersema DPM2, Nneka Meka DPM3

The Foot and Ankle Online Journal 11 (2): 1

A history of lower extremity ulceration increases the risk for further ulceration, infection, and subsequent amputation. The best predictor of amputation is a history of previous amputation. The transmetatarsal amputation is often performed as a secondary procedure when infection has recurred after a digital or partial ray amputation.  Patients with a digital or partial ray amputation are at a higher risk for a more proximal amputation such as a transmetatarsal amputation (TMA) or a below knee amputation (BKA). The primary objective of this study is to determine the time frame between a digital or a partial ray amputation to TMA or a BKA, with a secondary objective to focus on the co-morbidities of the research patients. Our aim is to study the time frame from initial to subsequent amputations, in order to identify a “critical period” in which increased intervention may be beneficial. The study design consists of a three-year retrospective chart review of patients who initially received a digital or partial ray amputation and subsequently required a TMA or a BKA. The median time from digital or partial ray amputation to transmetatarsal amputation was 55 days. The median time from digital or partial ray amputation to below knee amputation was 21 days. A possible explanation for the median number of days to BKA being shorter than the median number of days to TMA could be that the patients with more serious comorbidities were more likely to require a BKA emergently. The interventions during the critical period include tighter glucose control and lower extremity angiograms with close follow-up to address the underlying comorbidities of these patients.  Further research is needed regarding the degree of effectiveness of these interventions and their ultimate success in limb salvage.

Keywords: transmetatarsal amputation, below knee amputation, infection, digital amputation

ISSN 1941-6806
doi: 10.3827/faoj.2018.1102.0001

1 – Chief Resident, Department of Podiatry, Ochsner Health System, New Orleans, LA
2 – Second year resident, Department of Podiatry Ochsner Health System , New Orleans, LA
3 – Staff Physician, Department of Podiatry Ochsner Health System, New Orleans, LA
* – Corresponding author: carmen.bruno@ochsner.org


Diabetic patients are generally at a higher risk for developing non-healing ulcerations of the lower extremity. It is estimated that 15% of patients with diabetes will develop lower extremity ulcerations at some point in their lifetime [1]. The overall incidence of diabetic lower extremity amputations in the United States is 195 per 100,000 per year [2]. A few of the common diabetes-related risk factors for developing ulcerations include peripheral neuropathy, vascular disease, and a history of ulceration or amputation [3].

Microvascular and macrovascular disease are complications of diabetes. Hyperglycemia can lead to microvascular complications such as diabetic neuropathy, nephropathy, and retinopathy. Macrovascular disease can lead to complications including coronary artery disease and peripheral arterial disease (PAD). Peripheral arterial disease is commonly seen in diabetics and can result in prolonged healing of ulcerations secondary to arterial insufficiency. Additionally, PAD can lead to lack of oxygenation and difficulty in delivering antibiotics to the infection site, thus impairing the ability to resolve infections [4].

A history of a lower extremity ulceration increases the risk for further ulceration, infection and subsequent amputation. The best predictor of amputation is a history of previous amputation [4, 5]. The transmetatarsal amputation is often performed as a secondary procedure when infection has recurred after a digital or partial ray amputation.  Patients with a digital or partial ray amputation are at a higher risk for a more proximal amputation such as a TMA or a BKA [6].

To our knowledge, the period between the initial amputation to a more proximal amputation such as a TMA or a BKA has not been well studied. Our aim is to study the time frame from initial to subsequent amputations, in order to identify a “critical period” in which increased intervention may be beneficial.

The primary objective of this study is to determine the time frame between a digital or a partial ray amputation to TMA or a BKA, with a secondary objective to focus on the co-morbidities of the research patients. Co-morbidities related to the microvascular and macrovascular disease including end stage renal disease (ESRD) and PAD, respectively, will be included.

Methods

The study design consists of a three-year retrospective chart review of patients who initially received a digital or partial ray amputation who subsequently required a TMA or a BKA. After approval was obtained by the Ochsner IRB, the research data miner was provided with the procedure codes and inclusion criteria. The charts were then further analyzed to see if they met the inclusion criteria. The inclusion criteria are displayed in Figure 1. Data was analyzed utilizing quantitative methods to determine the time frame from a distal amputation to transmetatarsal amputation of the same limb. Data was also analyzed to determine the time frame from a digital or partial ray amputation to a BKA or TMA of the same limb. The comorbidities of diabetes, PAD, and ESRD from the research patients were also included in the study for further analysis. Statistical analysis was completed utilizing SAS version 9.4. Tests were performed with significance level of α=0.05.

Patients undergoing transmetatarsal amputation in the past 3 years of the same limb.
Patients undergoing a below knee amputation in the past 3 years of the same limb.

Figure 1 Inclusion criteria.

Results

From 2012 to 2015, there were 96 transmetatarsal amputations and 91 below knee amputations performed at Ochsner Clinic Foundation. From the patients with a history of transmetatarsal amputation, 41% (40) had previously undergone digital or partial ray amputation. From the patients with a history of below knee amputation, 25% (23) had previously undergone digital or partial ray amputation. Of the patients with a history of transmetatarsal amputation, 87% (35) had diabetes, 70% (28) had PAD and 57% (23) had ESRD. Of the patients with a history of a BKA, 86% (20) had diabetes, 86% (20) had PAD and 56% (13) had ESRD. This information is displayed in Figure 2.

For the patients with a history of BKA, the comorbidities of ESRD, PAD and diabetes were all found to be statistically significant risk factors with p-value of 0.0001, 0.0068 and 0.0058 respectively.

Figure 2 Comorbidities of BKA and TMA patients.

For patients with a history of TMA, the comorbidity of diabetes was found to be a statistically significant risk factor with a p-value of 0.00029. The comorbidities of ESRD and PAD were not found to be statistically significant risk factors in patients with a history of TMA. The median time from digital or partial ray amputation to transmetatarsal amputation was 55 days. The median time from digital or partial ray amputation to below knee amputation was 21 days. This information is displayed in Figure 3.

Discussion

A limitation of this study is that the complete list of comorbidities for each patient was not taken into account. Some patients may have had more severe comorbidities than others, leading to a skewed time frame between digital or partial ray amputation to TMA and BKA.  Additionally, the studied cohorts were not matched in terms of age, gender, and complete list of comorbidities as previously mentioned. There was no overlap between patients included in the TMA and BKA cohorts.

A possible explanation for the median number of days to BKA being shorter than the median number of days to TMA could be that the patients with more serious comorbidities were more likely to require a BKA emergently. However, the difference in days to a more proximal amputation was not statistically significant between the separate groups, with a p-value of 0.0709.

Figure 3 Median days to BKA and TMA.

Total Patients BKA Patients TMA Patients P-value Wilcoxon Test
N=63 N=23 N=40
Days to procedure, Median (q1-q5 Range) Median: 21

(13-79)

Median: 55

(19-204)

0.0709 Z = -1.8063

Figure 4 Median and range (q1-q5) of days to BKA and TMA.

Conclusion

Patients with a digital or partial ray amputation are at a higher risk for more proximal amputations such as a transmetatarsal amputation and below knee amputation. The average time frame from a distal amputation to a more proximal amputation could provide additional information regarding the “critical period” in which intervention is key to prevention. The “critical period” for this study refers to the median time frame from a distal amputation to a TMA or a BKA, which were 55 days and 21 days respectively. 90% of patients requiring a BKA had the procedure done within 79 days, and 90% of patients requiring a TMA had the procedure done within 204 days.

A conclusion that can be drawn from the data is that patients should be carefully monitored for at least two months after an initial digital or partial ray amputation. A more conservative approach, using the 90th percentile data, is to monitor patients for a longer time frame of up to seven months to reduce the incidence of more proximal amputations.

The interventions during the critical period include tighter glucose control and lower extremity angiograms with close follow-up to address the underlying comorbidities of these patients.  Further research is needed regarding the degree of effectiveness of these interventions and their ultimate success in limb salvage.

Funding Declaration

Financial Support for this project was provided from Ochsner Health System.

Conflict of Interest

None reported.

Acknowledgements

We would like to thank the research department at Ochsner Medical Center for their assistance with this project.

References

  1. Elsharawy MA. Outcome of midfoot amputations in diabetic gangrene. Ann Vasc Surg. 2011;25(6):778-82.
  2. Suckow BD, Newhall KA, Bekelis K, et al. Hemoglobin A1c Testing and Amputation Rates in Black, Hispanic, and White Medicare Patients. Ann Vasc Surg. 2016;36:208-217.
  3. Frykberg RG, Zgonis T, Armstrong DG, et al. Diabetic foot disorders. A clinical practice guideline (2006 revision). J Foot Ankle Surg. 2006;45(5 Suppl):S1-66.
  4. Borkosky SL, Roukis TS. Incidence of re-amputation following partial first ray amputation associated with diabetes mellitus and peripheral sensory neuropathy: a systematic review. Diabet Foot Ankle. 2012;3
  5. Fowler MJ. Microvascular and Macrovascular Complications of Diabetes. Clinical Diabetes 2008 Apr; 26(2): 77-82.
  6. Gambardella GV. Blume PA.  Understanding The Biomechanics of the Transmetatarsal Amputation. Podiatry Today. 2013;26(3):46-56.

Spring 2018

Issue 11 (1), 2018


Effects of a foot orthosis custom-made to reinforce the lateral longitudinal arch on three-dimensional foot kinematics
by Shintarou Kudo, Yasuhiko Hatanaka, Toshihiro Inuzuka


Conservative surgical management in an extreme diabetic foot case
by JM García-Sánchez, A Ruiz-Valls, A Sánchez-García, A Pérez-García


Choice of surgical treatment for patients with arthrosis of the ankle joint
by Kirill S. Mikhaylov, Vladimir G. Emelyanov, Alexandr Yu Kochish, Aleksandr A. Bulatov


A rare presentation of posterior compartment abscess in a diabetic patient: A case study
by Anthony Romano DPM/PGYIII, Kaitlyn L. Ward DPM/PGYIII, Byron Hutchinson, DPM FACFAS


A new technique using cruciate incisions for treating macrodactyly toe: Case report and review of the literature
by Mohammed Taifour Suliman MD FRCS


Cuboid navicular tarsal coalition: Presentation and evaluation with emphasis on magnetic resonance imaging appearance
by Angela Chang BS, Carly A. Lockard MS, Márcio B. Ferrari MD, Thomas O. Clanton MD, Charles P. Ho MD PhD


Eccrine syringofibroadenoma: A case report with dermatoscopic findings
by Mary A. Mooney MD and Myron A. Bodman DPM

Eccrine syringofibroadenoma: A case report with dermatoscopic findings

by Mary A. Mooney MD1* and Myron A. Bodman DPM1

The Foot and Ankle Online Journal 11 (1): 7

Eccrine syringofibroadenoma is a rare, benign adnexal lesion derived from cells of the acrosyringium of eccrine sweat glands.  This lesion has multiple clinical manifestations, including a reactive form, occurring in a pre-existing inflammatory or neoplastic dermatosis. Our case represents the typical presentation of the reactive form of eccrine syringofibroadenoma, including the dermoscopic appearance of the lesion.

Keywords: Eccrine syringofibroadenoma, dermoscopy, glomerular vessels, nummular eczema

ISSN 1941-6806
doi: 10.3827/faoj.2018.1101.0007

1 – College of Podiatric Medicine, Kent State University, 6000 Rockside Woods Blvd, Independence, OH 44131
* – Corresponding author: mmooney9@kent.edu


Pedal dermatological disorders are commonly limited to superficial fungal, viral and bacterial infections, pressure induced and genetic hyperkeratotic disorders, psoriasis and eczemas. Pedal neoplastic disease is much less common. There is a characteristic diagnostic delay of pedal neoplasms that can be attributed to the behavior of both patients and physicians. Patients may pay less attention to lower extremity problems, while in the primary care office the lower extremity physical examination may have a low priority. Physicians must also be aware of this complication of chronic dermatosis.

Case Report

A 62-year-old female presented with a 12-year history of intermittent nummular eczema on the right heel.  Treatment with triamcinolone 0.5% ointment had provided some relief (patient stated 20% improvement) but the lesion remained.  The patient wanted to know the diagnosis and accepted the risks and benefits of a punch biopsy.

The medical history was significant for epilepsy, treated with Dilantin. Physical examination demonstrated an erythematous hyperkeratotic plaque with fissures on the right heel measuring 1.5 cm. in diameter (Figure 1).  Dermoscopy examination with polarized light revealed enlarged irregular dermal vascular structures and opaque hyperkeratosis consistent with cutaneous neoplasia (Figure 2).

A 2-mm punch biopsy was performed and the specimen was sent for analysis.  The pathology diagnosis was reported as syringofibroadenoma. The patient was referred for a possible wide excision of the lesion.

Figure 1 Scaly plaque over lateral aspect of the right heel pad.

Figure 2 Dermoscopy image with polarized light detects glomerular dermal vessels partially obscured by opaque hyperkeratosis.

Figure 3 Thin anastomosing cords of epithelial cells surrounded by fibrovascular stroma (H&E x 10).

Discussion  

Eccrine syringofibroadenoma (ESFA) is a rare, benign  neoplasm of the acrosyringium of eccrine sweat glands, first described by Mascaro in 1963 [1].    ESFA has multiple clinical presentations. These were classified by Starink into four subtypes [2].  French added a fifth subtype, reactive [3]. The current classification system is: 1) solitary ESFA, non-hereditary, 2) multiple ESFA in hidrotic ectodermal dysplasia (Schöpf syndrome), 3) multiple ESFA without cutaneous findings (eccrine syringofibroadenomatosis), 4) nonfamilial unilateral linear ESFA (nevoid), and 5) reactive ESFA associated with inflammatory or neoplastic dermatoses [3].

This lesion typically presents in adult individuals, usually over the age of 40.  It frequently appears in the distal extremities. The reactive form is present in patients with inflammatory or neoplastic lesions, or in patients with peripheral neuropathy [4].  In these cases, eccrine syringofibroadenoma may arise in response to repetitive damage with subsequent repair of eccrine ducts [3]. The exact nature of the lesion, however, is not completely understood.  It may be hyperplastic, hamartomatous, or neoplastic[2]. Although ESFA typically has a benign course, it has been reported as a precursor to squamous cell carcinoma and pseudocarcinomatous hyperplasia [5, 6].  Malignant transformation of ESFA is rare, but has also been reported [7]. In these cases, it can be difficult to determine if the EFSA was the primary lesion, with a carcinoma developing within it, or if the EFSA developed in response to a previously existing cancer.

Clinically, ESFA presents as a slow growing, exophytic lesion with a verrucous or spongy, moist mosaic, or erythematous scaly plaque appearance [8].  The lesions may be solitary or multiple. It is frequently located on the extremities, with multiple case reports involving the foot and ankle, although other parts of the body such as the hand, wrist, finger, and lip, can be involved [2].

The dermoscopy image detected vascular structures consistent with glomerular vessels.  The structural pattern of vessels in dermoscopy play a critical role in the diagnosis of non-pigmented skin lesions [9].  Glomerular vessels are large-caliber reddish dots formed by tortuous capillaries curled up into a ball, resembling the glomerular apparatus of the kidneys and have been observed in a variety of neoplasms including Bowen’s disease, eccrine poroma, basal cell carcinoma, Merkel cell carcinoma as well as actinic keratoses and stasis dermatitis but have not been previously reported in ESFA [9].

The histologic appearance of the lesion demonstrates thin, anastomosing strands of epithelial cells, which can appear as cords, surrounded by a fibrovascular stroma. These strands are contiguous with the epidermis [8].  Eccrine syringofibroadenoma stains positive with epithelial membrane antigen and carcinoembryonic antigen[8]. In spite of the highly variable clinical presentation, the histologic appearance of ESFA is uniform for the different subtypes [3].  

Differential diagnosis may include prurigo nodularis, granulomatous dermatitis, fungal and bacterial infections, eccrine poroma, reticulated seborrheic keratosis, lichen sclerosis, and fibroepithelioma of Pinkus and amelanotic melanoma [8, 9].

Our patient represented a case of reactive eccrine syringofibroadenoma.  The patient had a 12-year history of intermittent nummular eczema on the right heel with only a partial response to topical steroids.  This type of clinical history is similar to other reported cases of reactive EFSA. Histologically, the lesion demonstrated the typical appearance of EFSA, with thin anastomosing cords of epithelial cells surrounded by fibrovascular stroma contiguous with the epidermis.  There were no signs of atypia or malignant transformation (Figure 3).

Our patient was referred for wide excision of the lesion to a plastic surgeon and to a dermatologist for a full body examination to search for additional lesions. Wide excision has been the mainstay of treatment [10], although cryotherapy [11], radiation [12], and carbon dioxide laser [13] are alternatives.  For early lesions, close observation and follow up may be an alternative to complete excision, especially if complete excision is difficult due to the size or location of the lesion [10]. The patient was lost to follow-up.

Acknowledgement

We would like to acknowledge the assistance of Ashfaq A. Marghoob MD, Director of Clinical Dermatology at Memorial Sloan Kettering Cancer Center, New York, for his help in interpreting the dermoscopy image

This case was written with the approval of the Kent State University Institutional Review Board.

References

  1. Mascaro JM. Considerations on fibro-epithelial tumors. Exocrine syringofibroadenoma. Ann Dermatol Syphiligr (Paris) 1963;90:143-53.
  2. Starink TM. Eccrine syringofibroadenoma: multiple lesions representing a new cutaneous marker of the Schöpf syndrome, and solitary nonhereditary tumors. J Am Acad Dermatol 1997;36(4):569-76.
  3. French LE. Reactive eccrine syringofibroadenoma: an emerging subtype. Dermatology 1997;195(4):309-10.
  4. Sirikham T, Rojhirunsakool S, Vachiramon V. Reactive Eccrine Syringofibroadenoma Associated with Neuropathy, Venous Stasis, and Diabetic Foot Ulcer. Case Rep Dermatol 2016;8(2):124-9.   
  5. Bjarke T, Ternesten-Bratel A, Hedblad M, Rausing A. Carcinoma and eccrine syringofibroadenoma: a report of five cases. J Cutan Pathol 2003;30(6):382-92.
  6. Kacerovska D, Nemcova J, Michal M, Kazakov DV. Eccrine syringofibroadenoma associated with well-differentiated squamous cell carcinoma. Am J Dermatopathol 2008;30(6):572-4.
  7. Katane M, Akiyama M, Ohnishi T, Watanabe S, Matsuo I. Carcinomatous transformation of eccrine syringofibroadenoma. J Cutan Pathol 2003;30(3):211-4.
  8. Lowell DL, Salvo NL, Weily WJ, Swiatek M, Sahli H. Multiple Eccrine Syringofibroadenoma of Mascaro of the Lower Extremity. J Am Podiatr Med Assoc 2016;106(6):433-8.
  9. Tiwary AK, Firdous J, Mishra DK, Chaudhary SS. A case report of reactive solitary eccrine syringofibroadenoma. Indian Dermatol Online J 2017;8(1):35-8.
  10. Cho E, Lee JD, Cho SH. A case of reactive eccrine syringofibroadenoma. Ann Dermatol 2011;23(1):70-2.
  11. Ozkaya DB, Su O, Bahalı AG, Topukçu B, Dizman D, Tosuner Z, et al. Solitary Eccrine Syringofibroadenoma and Successful Treatment with Cryotherapy. J Am Podiatr Med Assoc 2016;106(3):237-8.
  12. Morganti AG, Martone FR, Macchia G, Carbone A, Massi G, De Ninno M, et al. Eccrine syringofibroadenoma radiation treatment of an unusual presentation. Dermatol Ther 2010;23 Suppl 1:S20-3.
  13. Athanasiadis GI, Bobos M, Pfab F, Athanasiou E, Athanasiadis IE. Eccrine syringofibroadenoma treated with carbon dioxide laser. Clin Exp Dermatol 2009;34(2):261-3.

Cuboid navicular tarsal coalition: Presentation and evaluation with emphasis on magnetic resonance imaging appearance

by Angela Chang BS1, Carly A. Lockard MS1, Márcio B. Ferrari MD1, Thomas O. Clanton MD1,2, Charles P. Ho MD PhD1,*

The Foot and Ankle Online Journal 11 (1): 6

Tarsal coalition is an uncommon condition that is often overlooked in the adult population. The potential sequelae of untreated tarsal coalition include changes in gait mechanics, flattening of the longitudinal arch and degenerative changes in the hindfoot joints. Tarsal coalition should be considered as a possible diagnosis in the active younger patient who presents with frequent hindfoot pain and ankle sprains. Our report presents a case of an incidental finding of an asymptomatic cuboid-navicular tarsal coalition in a patient who presented with persistent Achilles tendinosis and reviews the available literature regarding this condition.

Keywords: cubonavicular tarsal coalition, cuboid-navicular coalition, tarsal coalition, Achilles tendinosis, MRI

ISSN 1941-6806
doi: 10.3827/faoj.2018.1101.0006

1 – Steadman Philippon Research Institute, Vail, Colorado, USA
2 – The Steadman Clinic, Vail, Colorado, USA
* – Corresponding author: charles.ho@sprivail.org


Tarsal coalition, thought to have first been described in the literature in 1877 [1], is an anatomical anomaly in which a fibrous, cartilaginous, or osseous coalition forms between two or more tarsal bones [2]. This condition occurs in less than 1% of the general population [3]. Approximately 48% of tarsal coalitions are talocalcaneal, 44% are calcaneonavicular, and the remaining 8% occur in other tarsal joints [3,4]. It has been suggested that tarsal coalition occurs during embryonic development due to failure of mesenchymal differentiation and segmentation, and may be genetic in etiology [5,6]. Tarsal coalition is found in association with pes planus, hindfoot rigidity and peroneal spastic flatfoot [7-9].

Imaging evaluation of suspected tarsal coalition should begin with anteroposterior (AP), 45-degree internal oblique, and lateral foot radiographs with the AP and lateral taken weight bearing [10].  These three views are typically sufficient for diagnosis of calcaneonavicular and talonavicular coalition. On radiographs, calcaneonavicular coalition is best visualized on a 45-degree internal oblique, and can present as an “anteater nose sign” on the lateral view [10]. Talocalcaneal coalitions may be difficult to visualize directly due to the complex orientation of the subtalar joint, but are associated with secondary signs such as talar beaking, narrowing of the posterior subtalar joint with poor visualization of the middle facets, rounding of the lateral talar process, and “C sign” created by bone bridging between the talar dome and sustentaculum tali [10].

Additional computerized tomography (CT) and magnetic resonance imaging (MRI) are recommended to visualize the size, location and extent of joint involvement more accurately [11-13]. CT should be reconstructed in both the coronal and axial planes. Non-osseous coalitions may appear as joint space narrowing and marginal reactive bone changes [10]. MRI in the coronal and sagittal planes is recommended to allow differentiation between osseous, cartilaginous, and fibrous bridges [10].

Case Report

A 23-year-old man began experiencing heel pain three years prior to presentation at our office. His past medical history was negative for diabetes mellitus, traumatic or chronic foot and ankle injuries, or rheumatologic disease. Three years prior to presentation at our clinic the patient experienced acute onset of left heel pain focused around the medial aspect of the distal Achilles following a high-elevation mountain hike. This pain improved with eccentric calf exercises and gentle stretching. However, another alpine hiking activity one year later resulted in onset of pain in the right heel, which did not improve satisfactorily with conservative treatment including gel heel inserts, calf raises and inversion-eversion exercises. The patient began to experience chronic recurrent bilateral distal Achilles pain that varied between 0 and 9 points on a 10-point scale. The patient did not experience any other midfoot or hindfoot pain. The pain was exacerbated with running and improved with rest and diclofenac 1% gel (Voltaren Topical). Four days prior to presentation at our clinic the patient underwent a right hip arthroscopy for femoroacetabular impingement, including labral repair and acetabular and femoral osteochondroplasty.

Upon physical exam, the patient was wearing a hip brace and limited to 20 pounds of flatfoot weight-bearing on the right and therefore standing alignment could not be assessed. Bilaterally there was swelling but no tenderness to palpation along the Achilles tendon at the insertion, over the lateral calcaneal ridge, or over the lateral or medial aspect of the tendon. Heel raises were not performed due to his postoperative right hip arthroscopy status. Range of motion of the right ankle with knee straight showed dorsiflexion to 0°, plantarflexion to 40°, inversion to 15°, eversion to 10°, adduction of 15°, and abduction of 10°.  Left ankle dorsiflexion with the knee bent was to 10°.  Range of motion was equal bilaterally. The patient had 5/5 manually-tested plantarflexion strength bilaterally.

Bilateral calcaneal lateral and axial radiographs were obtained.  There was no evidence of Haglund’s deformity, calcifications, or fracture. Sagittal, coronal and axial magnetic resonance images of both ankles were also obtained.

On MRI of the right ankle, mild longitudinal thickening of the Achilles tendon with increased signal and tendinosis of the distal tendon was visualized. No focal tear defect or retraction was identified. A mild retrocalcaneal bursitis was noted. The cuboid-navicular tarsal coalition was identified on the MRI.  The coalition appeared as an osseous prominence of the cuboid-navicular junction, with prominent narrowing of the cuboid-navicular bone junction, and sclerosis, irregularity, thinning, slight pitting, and appearance of interdigitation of the bone margins at the junction. These characteristics are visible both on sagittal (SAG) and coronal non-fat-suppression (COR non-FS) and fat-suppression (FS) images. (Figures 1 and 2). Surrounding bone showed poorly defined increased signal compatible with stress-related edema; this increased signal is most apparent on the FS images (Figures 1b and 2b).   Stress-related change and edema were also visualized in the surrounding soft tissue; this increased signal is most apparent on the FS images (Figures 1b and 2b). The cuboid-navicular tarsal coalition appearance, with the narrowing of the bone junction and appearance of bone margin interdigitation and thinning but lack of complete cortical and trabecular bone continuity and marrow bridge, is typical of a soft tissue fibro-cartilaginous coalition or mixed osseous/soft tissue coalition.

Figure 1 Sagittal proton density-weighted turbo spin echo non-fat-suppressed (A) and fat-suppressed (B) magnetic resonance image showing coalition (arrow). Severe narrowing of the cuboid-navicular junction is seen with opposing bone margin irregularity, sclerosis, appearance of interdigitation thinning and pitting. Bone and surrounding soft tissue increased signal and stress-related edema are more apparent and prominent on the fat-suppressed image.

Figure 2 Coronal proton density-weighted turbo spin echo non-fat-suppressed (A) and fat-suppressed (B) magnetic resonance image showing coalition (arrow). Severe narrowing of the cuboid-navicular junction is seen with opposing bone margin irregularity, sclerosis, appearance of interdigitation thinning and pitting. Bone and surrounding soft tissue increased signal and stress-related edema are more apparent and prominent on the fat-suppressed image.

On the left side there were similar signs of Achilles tendinosis and retrocalcaneal bursitis. No tarsal coalition was present on the left side. The patient was made aware of the incidental finding and the potential for the coalition to become symptomatic in the future. The patient deferred treatment of the coalition and elected to manage the Achilles tendinosis conservatively.

Discussion

Calcaneonavicular and talocalcaneal tarsal coalitions are extensively reported in the literature. However, a much smaller number of cuboid-navicular cases have been reported, with varying symptoms at presentation.  Current literature suggests that this type of coalition is often asymptomatic, with exacerbation of pain and/or peroneal spastic flatfoot only with increased activity [14]. Del Sel and Grand reported the diagnosis of bilateral cuboid-navicular coalitions in a 45-year-old patient who underwent radiographic examination due to trauma to both feet [15].  Chu et al. also reported an incidental finding of a cuboid-navicular coalition upon CT evaluation in a 35-year-old patient following a comminuted pilon fracture [16].

In addition to reports on asymptomatic coalitions, several authors have reported symptomatic coalitions, with symptoms including dorsal and midfoot pain and decreased range of motion [13,14,17,18]. Awan et al. reported a case on a 17 year old patient with chronic unremitting pain at rest that was exacerbated during sports-related activities [14]. Feliu reported a case in which the only symptom was two years of intermittent pain in the dorsum of the foot with otherwise normal findings upon physical exam, while Johnson et al. reported a case on a patient with fixed pes planus deformity presented with midfoot pain associated with a decrease in subtalar and transverse tarsal joint motion [18,3].  

The patient age at the time of diagnosis varies from 9 [17] to 45 [15] years old. We found a comparable number of studies reporting male [3,14,15,18,19] and female [4,16,17,20] patients. However, the reported time from the beginning of the symptoms to the diagnosis ranged from two weeks [19]  to several years [3,13,17]. Delayed diagnosis of cuboid-navicular coalitions is often reported in the literature due to missed coalition on radiographic exam. Routine images often show minimal changes with an absence of secondary signs that are seen with other tarsal coalitions and findings specific to cuboid-navicular coalitions [14].

A high level of suspicion must be present in order to correctly diagnose cuboid-navicular coalition, as the only radiographic finding may be an abnormal relationship between the posterior medial cuboid and plantar lateral navicular [14]. Cuboid-navicular coalition does not cause talar beaking, so the absence of the talar beak sign cannot be used to exclude the presence of this type of coalition [14]. Although some case studies reported successful diagnosis using radiographs, [18,19,21] several authors reported negative radiographic findings [3,4].

Fibrocartilaginous and bony forms of cuboid-navicular coalition exist and MRI can be useful in distinguishing between them [3,13,14,16,17]. MRI findings in the context of a bony coalition include a continuous marrow across the coalition bridge [10]. MRI may be superior to CT for the diagnosis of fibrous coalitions as the MRI allows better differentiation between bony and fibrous changes [10]. Furthermore, MRI proton density fat-suppressed (PD FS), T2-weighted fat-suppressed (T2 FS), and short T-1 inversion recovery (STIR) images allow visualization of reactive edema [10]. Different stages of coalition ossification may exist in a single patient. For example, Del Sel and Grand and Piqueres et al. reported cases of bilateral cuboid-navicular coalition, with an osseous coalition on one side and a cartilaginous coalition on the contralateral side [15,21].

The treatment for symptomatic cuboid-navicular coalition varies from conservative [14,18] to surgical treatment [3,13,17,20]. Awan et al reported successful treatment using physical therapy, although the reported follow-up was only 3 months [14]. Several authors have reported their surgical treatment outcomes, with generally good results. Johnson et al. reported failure of conservative treatment including short leg cast immobilization, ultimately progressing to surgical treatment, with the resection of the osseous bar. This was performed through a single lateral curvilinear incision from the inferior aspect of the lateral malleolus to the distal medial aspect of the navicular [3]. Piqueres et al also reported good results following surgical treatment, with 1 year follow-up, and return to previous sports participation with no recurrence of pain [21]. During surgical treatment the resected bone surfaces may be separated using bone wax [4], adipose tissue [13], or the extensor digitorum brevis muscle [17] to avoid coalition recurrence. However, Hounshell  reported that donor site morbidity and recipient instability can occur with the use of wax or muscle belly as the interposition spacer and suggested the use of an acellular human dermal regenerative tissue matrix as an acceptable alternative since it was associated with good stability and no regeneration of the bone bar [20]. To our knowledge, the largest surgically-treated case series reported is from Sarage et al., which included four patients aged 15 to 35 years with fibrous coalition who were surgically treated with coalition resection and adipose tissue interposition with good results [13].

Conclusion

The purpose of this case study is to present the radiographic and MRI findings in a patient with asymptomatic cuboid-navicular coalition in the context of symptomatic Achilles tendinosis. MRI is an important diagnostic component of the evaluation of tarsal coalition, especially in the less commonly affected tarsal bones.

Funding declaration

No funding was received for this work.

Conflict of interest declaration

All authors: Steadman Philippon Research Institute Research Support from: Smith & Nephew Endoscopy, Arthrex, Siemens Medical Solutions, USA, Ossur Americas, Vail Valley Medical Center.

Charles P. Ho: Steadman Philippon Research Institute (Research Advisory Committee), Rotation Medical (Consultant).

Thomas O. Clanton: Arthrex, Inc. (Consultant/speaker fees, and royalties and in-kind donations of surgical supplies for research), Stryker, Inc. (Consultant/speaker fees and royalties), Steadman Philippon Research Institute (Research Advisory Committee).

References

  1. Moraleda L, Gantsoudes GD, Mubarak SJ. C Sign: Talocalcaneal coalition or flatfoot deformity? J Pediatr Orthop 2014;34:814–9.
  2. Kulik SA, Jr., Clanton TO. Tarsal coalition. Foot Ankle Int 1996;17(5):286-96.
  3. Johnson TR, Mizel MS, Temple T. Cuboid-navicular tarsal coalition — presentation and treatment: A case report and review of the literature. Foot Ankle Int 2005;26(3):264-6.
  4. Kamiya T, Watanabe K, Teramoto A, Yamashita T. Cuboid-navicular tarsal coalition in an adolescent female athlete: A case report. JBJS Case Connect, 2015 Nov 11; 5 (4): e93.
  5. Leonard MA. The inheritance of tarsal coalition and its relationship to spastic flat foot. J Bone Joint Surg Br 1974;56b(3):520-6.
  6. Herzenberg JE, Goldner JL, Martinez S, Silverman PM. Computerized tomography of talocalcaneal tarsal coalition: A clinical and anatomic study. Foot Ankle 1986;6(6):273-88.
  7. Anderson RJ. The presence of an astragalo-scaphoid bone in man. J Anat Physiol 1880;14(Pt 4):452-5.
  8. Harris RI, Beath T. Etiology of peroneal spastic flat foot. J Bone Joint Surg Br 1948;30-B(4):624-34.
  9. Badgley CE. Coalition of the calcaneus and the navicular. Arch Surg 1927;15:75-88.
  10. Newman JS, Newberg AH. Congenital tarsal coalition: multimodality evaluation with emphasis on CT and MR imaging. Radiographics 2000;20(2):321-32; quiz 526-7, 32.
  11. Lawrence DA, Rolen MF, Haims AH, Zayour Z, Moukaddam HA. Tarsal coalitions: Radiographic, CT, and MR imaging findings. HSS J 2014;10(2):153-66.
  12. Lemley F, Berlet G, Hill K, Philbin T, Isaac B, Lee T. Current concepts review: Tarsal coalition. Foot Ankle Int 2006;27(12):1163-9.
  13. Sarage AL, Gambardella GV, Fullem B, Saxena A, Caminear DS. Cuboid-navicular tarsal coalition: Report of a small case series with description of a surgical approach for resection. J Foot Ankle Surg 2012;51(6):783-6.
  14. Graham JA, Awan O. The rare cuboid-navicular coalition presenting as chronic foot pain. Case Rep Radiol 2015:1-4.
  15. Del Sel JM, Grand NE. Cubo-navicular synostosis; a rare tarsal anomaly. J Bone Joint Surg Br 1959;41-b(1):149.
  16. Chu JS, Underriner T, Yegorov A. A rare case of cubonavicular coalition. Radiol Case Rep.
  17. Prado MP, Mendes AA, Olivi R, Amodio DT. Cuboid-navicular tarsal coalition. Rev Bras Ortop 2010;45(5):497-9.
  18. Feliu EC. Cubonavicular synostosis. A case report. Acta Orthop Belg 1991;57(3):306-8.
  19. Waugh W. Partial cubonavicular coalition as a cause of peroneal spastic flat foot. J Bone Joint Surg Br 1957;39-b(3):520-3.
  20. Hounshell CR. Regenerative tissue matrix as an interpositional spacer following excision of a cuboid-navicular tarsal coalition: A case study. J Foot Ankle Surg 2011;50(2):241-4.
  21. Piqueres X, de Zabala S, Torrens C, Marin M. Cubonavicular coalition: A case report and literature review. Clin Orthop Relat Res 2002(396):112-4.