Tag Archives: external fixation

Staged surgical intervention in the treatment of septic ankle arthritis with autologous circular pillar fibula augmentation: A case report

by Sham J. Persaud DPM, MS1*; Colin Zdenek DPM2; Alan R. Catanzariti DPM3

The Foot and Ankle Online Journal 10 (3): 6

Surgical management of chronic septic arthritis of the ankle joint is a challenging problem. Failure to initiate appropriate antibiotic therapy and perform incision and drainage within the first 24 to 48 hours of onset can result in subchondral bone loss and permanent joint dysfunction. Patients with chronic infection are not only at risk for loss of joint function, but also limb loss. This case report presents a staged procedure for limb salvage of patients with chronic septic arthritis of the ankle joint. Our technique includes use of both internal and external fixation, along with infection control and autologous pillar grafts. Though our case study is limited, the results are comparable to previous studies. This approach appears to be reasonable for limb salvage in end-stage degenerative joint disease following septic ankle arthritis.

Keywords: Septic arthritis, ankle, pillar graft, internal fixation, external fixation

ISSN 1941-6806
doi: 10.3827/faoj.2017.1003.0006

1 – West Penn Hospital, The Foot and Ankle Institute, Pittsburgh, PA 15224
2 – Silicon Valley Reconstruction Foot and Ankle Fellow, Palo Alta Medical Foundation, Mountain View, CA 94040
3 – Director of the Residency Training Program, West Penn Hospital, The Foot and Ankle Institute, Pittsburgh, PA 15224
* – Corresponding author: Sham Persaud, shamjoseph.persaud@ahn.org


Surgical management of chronic septic arthritis of the ankle joint is a challenging problem. Failure to initiate appropriate antibiotic therapy and perform incision and drainage within the first 24 to 48 hours of onset can result in subchondral bone loss and permanent joint dysfunction. Joint function after Staphylococcus aureus (S. aureus) septic arthritis is generally lost 25-50% of the time [1-4]. The mortality rate for septic arthritis has been reported as high as of 10-15% [1-2, 5-8].

Internal ankle arthrodesis techniques are reported to have between 88% to 100% primary fusion rates in patients with aseptic arthritis [9-12]. However the fusion rate for ankle arthrodesis in the setting of sepsis is roughly 71% to 93% [13-19].

Surgical management of septic arthritis requires debridement of all non-viable infected soft tissue and bone in order to eradicate infection [14-15, 20]. In addition to debridement, the use of local antibiotic delivery through polymethylmethacrylate (PMMA) has been shown to be an effective adjunct in treating infection [21-24]. Bactericidal levels of antibiotics from PMMA spacers are achieved through the process of elution where high concentrations of antibiotic are released locally, with minimal systemic effect, and limited risk to the patient. Peak antibiotic concentrations are mostly reached within the first week after placement; however, some studies have shown antibiotics may still be released at effective concentrations even after 4-6 weeks of implantation [25-30].

The use of long term intravenous (IV) or suppressive oral antibiotic therapy in conjunction with debridement is an important part of treatment. A 2-6 week course of IV antibiotic therapy is recommended depending on the severity of the infection and host immunity [14, 15, 20, 31].   

Fixation techniques for arthrodesis of the diseased ankle secondary to septic arthritis have been controversial. External fixation has been shown to provide adequate torsional stability, but is less effective in maintaining sagittal plane stability. On the other hand, internal fixation has been shown to provide excellent sagittal plane stability, but limited torsional stability. [19] Some authors believe a combination of both fixation techniques lead to optimal outcomes [12, 14, 15, 19, 20].

A concern with arthrodesis is following septic arthritis is loss of limb length. Cancellous bone graft has been shown to be effective in aiding with small defects [15, 20, 31]. Free vascularized bone graft has also been shown to be effective with large bony defects [14, 15, 20]. Use of allograft or synthetic bone grafts have rarely been mentioned in the literature [32]. One technique which has been described in aseptic ankle joint arthrodesis is the use of fibular pillar grafts as structural grafts to maintain length [33].

Patients with chronic infection are not only at risk for loss of joint function, but also limb loss. Cierny et al. related a 25% amputation rate for patients with arthrodesis of septic ankle joints [15]. This case report presents a staged procedure for limb salvage of patients with chronic septic arthritis of the ankle joint.

Case Report

A 54-year-old female with chronic right septic ankle arthritis for 6 months presented for evaluation. The patient had undergone arthrocentesis with corticosteroid, I&D with washout and long-term IV antibiotic therapy. She was offered a below knee amputation elsewhere but was reluctant to proceed and sought a second opinion. Her pre-operative radiographs can be seen in Figure 1 A-C and pre-operative MRI may be seen in Figure 2 A-B.  The patient chose to proceed with staged surgical approach for limb salvage.

Figure 1 Pre-operative radiographs; Mortise view, AP view, and Lateral view.

Figure 2 Pre-operative MRI; T1 Sagittal view, T2 Sagittal view.

The patient underwent needle biopsy of the tibia and talus with arthroscopic debridement. Arthroscopy was performed in standard fashion using a 2.7mm 30-degree arthroscope, utilizing a burr and shaver for ankle joint debridement.  Arthroscopic evaluation of the ankle joint revealed destruction of both tibial and talar articular surfaces.  Cartilage of both articular surfaces was degraded and granular in nature.  Cultures recovered S. aureus infection of the tibia.

Thirteen days later, open arthrotomy of the ankle joint with extensive debridement of the tibia and talus, as well as insertion of a Vancomycin cement spacer was performed.  The arthrotomy was performed using a lateral approach with a fibular osteotomy. The fibula was sent for pathology evaluation and culture, which were shown to be free of any bacterial infection.  Debridement was performed through osteotomies of both the tibia and talus which included the articular cartilage and subchondral plate (Figure 3). The joint was then pulse lavaged with 3L of normal saline-bacitracin mixture and a Vancomycin PMMA spacer was placed within the current ankle joint (Figure 4). This was then stabilized with a monolateral external fixator. The patient was placed on 6 weeks of antibiotic therapy by Infectious Disease including IV Cephazolin and PO Rifampin.

Figure 3 Intra-op radiograph status post fibula take down and wide excisional debridement of tibia and talus.

Figure 4 Intra-op radiographs and picture of Vancomycin PMMA spacer.

Ten weeks later, the patient underwent intramedullary (IM) nail tibiotalocalcaneal arthrodesis (TTC) (Figure 5). The original lateral incision was utilized to access the ankle joint.  The Vancomycin spacer was removed and soft-tissue specimens from the tibia and talus, which were sent for frozen section evaluation by pathology, were negative for infection. The bony surfaces were then prepared for arthrodesis in standard fashion using curettes, osteotomes, and drills. The subtalar joint was prepared in a similar fashion. The ankle was grafted with morselized femoral head combined with bone morphogenic proteins to provide osteoconduction and osteoinduction, as well as fibular pillar grafts to provide structural support and maintain length.  Fixation was accomplished with an IM nail. The patient remained non-weight bearing for 3 months.  She was then transitioned into a fracture boot for an additional month and then into a sneaker. No major or minor complications were noted throughout her recovery process. The patient has continued to improve throughout the post-operative course and is able to bear weight without assistance in standard foot gear. Serial radiographs have demonstrated complete union of all involved joints (Figure 6).

Figure 5 Intra-op radiographs (axial view, AP view, and lateral view) of IM nail with fibular pillar grafts.

Figure 6 Final radiographs showing consolidation (AP ankle, oblique ankle, lateral view) of IM nail with fibular pillar grafts.

Discussion

The fusion rate within the literature varies dramatically. Hawkins showed a variation between 71-94% depending on the control of infection within the joint [13]. Richter also reported a fusion rate of 86.6% for septic ankles [14]. Cierny et al reported results of 83% to 100%. Cierny believed this was secondary to the quality of the surrounding soft tissues. These cases used either external, or hybrid fixation techniques for their fusion [15].

Treatment of S. aureus septic ankle arthritis should include immediate lavage and debridement of the joint with culture and sensitivity driven antibiotic therapy [14, 15, 20].  However, this treatment alone leaves the patient predisposed to continued pain and discomfort secondary to sequela of septic arthritis. Therefore, ankle arthrodesis should be considered as a long-term option following resolution of the infection [4].

External fixation or a hybrid of external and internal fixation has been recommended for arthrodesis following septic ankle arthritis. We used a solitary IM nail for fixation in our cases. Klouche et al. discussed the use of internal fixation in a one-stage procedure using two cross screws through a lateral approach. There technique provided a cure rate of 85% and a consolidation rate of 89.5% at 4.8 months. Empiric antibiotics were administered to all patients and were modified based on culture and sensitivity results obtained at the time of surgery. No local antibiotics were used with their technique [34]. We used IV antibiotics before and after our definitive procedure, as well as, a Vancomycin loaded cement spacer following debridement of the infected bone.

Though our case study is limited, the results have been comparable to previous studies. This approach appears to be reasonable for limb salvage in end-stage degenerative joint disease following septic ankle arthritis. An evidence based study with increased numbers of patients and long term follow up would be beneficial in further accessing this technique for the treatment of septic arthritis of the ankle.

 

References

  1. Cooper C, Cawley MID. Bacterial arthritis in an English health district: a 10 year review. Ann Rheum Dis. 1986; 45:458-463.
  2. Peters RHJ, Rasker JJ, Jacobs JWG, Prevo RL, Karthaus RP. Bacterial arthritis in a district hospital. Clin Rheumatol. 1992; 11:351-355.
  3. Youssef PP, York JR. Septic arthritis: a second decade of experience. Aust N Z J Med. 1994; 24:307-311.
  4. Kaandorp CJE, Krunen P, Bernelot Moens HJ, Habbema JDF, Van Schaardenburg D. The outcome of bacterial arthritis: A prospective community-based study. Arthritis Rheum. 1997; 40(5):884-92.
  5. Meijers KAE, Dijkmans BAC, Hermans J, van den Broek PJ, Cats A. Non-gonococcal infectious arthritis: a retrospective study. J Infect. 1987; 14:13-20.
  6. Yu LP, Bradley JD, Hugenberg ST, Brandt KD. Predictors of mortality in non-postoperative patients with septic arthritis. Scand J Rheumatol. 1992; 21:142-144.
  7. Mathews C. J., Weston V. C., Jones A., Field M., Coakley G. Bacterial septic arthritis in adults. The Lancet.2010; 375(9717):846-855.
  8. Miller A, Abduljabbar F, Jarzem P. Polyarticular Septic Arthritis in an Immunocompetent Adult: A Case Report and Review of the Literature. Case Rep Orthop. 2015; 2015:602137.
  9. Scranton PE Jr. Use of internal compression in arthrodesis of the ankle. J Bone Joint Surg Am. 1985; 67:550–555.
  10. Mann RA, Rongstad KM. Arthrodesis of the ankle: a critical analysis. Foot Ankle Int. 1998; 19:3–9.
  11. Zwipp H, Grass R, Rammelt S, Dahlen C. Arthrodesis: non-union of the ankle—arthrodesis failed. Chirurg. 1999; 70:1216–1224.
  12. Kollig E, Esenwein SA, Muhr G, Kutscha-Lissberg F. Fusion of the septic ankle: experience with 15 cases using hybrid external fixation. J Trauma. 2003 Oct;55(4):685-91.
  13. Hawkins BJ, Langerman RJ, Anger DM, Calhoun JH. The Ilizarov technique in ankle fusion. Clin Orthop. 1994; 303:217–225.
  14. Richter D, Hahn MP, Laun RA, Ekkernkamp A, Muhr G, Ostermann PA. Arthrodesis of the infected ankle and subtalar joint: technique, indications, and results of 45 consecutive cases. J Trauma. 1999; 47:1072–1078.
  15. Cierny G III, Cook WG, Mader JT. Ankle arthrodesis in the presence of ongoing sepsis: indications, methods, and results. Orthop Clin North Am. 1989;20:709–721.
  16. Johnson EE, Weltmer J, Lian GJ, Cracchiolo A III. Ilizarov ankle arthrodesis. Clin Orthop. 1992; 280:160–169.
  17. Lonner JH, Koval KJ, Golyakhovsky V, Frankel VH. Posttraumatic nonunion of the distal tibial metaphysis: treatment using the Ilizarov circular external fixator. Am J Orthop. 1995; suppl: 16–21.
  18. Stasikelis PJ, Calhoun JH, Ledbetter BR, Anger DM, Mader JT. Treatment of infected pilon nonunions with small pin fixators. Foot Ankle. 1993; 14:373–379.  
  19. Thordarson DB, Patzakis MJ, Holtom P, Sherman R. Salvage of the septic ankle with concomitant tibial osteomyelitis. Foot Ankle Int. 1997; 18:151–156.  
  20. Cierny G, Zorn EZ. Arthrodesis of the tibiotalar joint for sepsis. Foot Ankle Clin 1996; 1:177– 97.  
  21. Chen NT, Hong HZ, Hooper DC, May JW. The effect of systemic antibiotic and antibiotic impregnated polymethylmethacrylate beads on the bacterial clearance in wounds containing contaminated dead bone. Plastic Reconst Surg 1993; 97(2):1305–11.
  22. Donati D, Biscaglia R. The use of antibiotic impregnated cement in infected reconstructions after resection for bone tumours. J Bone Joint Surg Br 1998; 80(6):1045– 50.
  23. Popham GJ, Mangino P, Seligson D, et al. Antibiotic impregnated beads: Part II: factors in antibiotic selection. Orthop Rev 1991; 20:331–7.
  24. Ostermann PA, Henry SL, Seligson D. The role of local antibiotic therapy in the management of compound fractures. Corr 1993; 295:102– 11.
  25. Antrum RM, Solomkin JS. A review of antibiotic prophylaxis for open fractures. Orthop Rev 1987; 16:81–9.
  26. Eckman JB Jr, Henry SL, Manginio PD, Seligson D. Wound and serum levels of tobramycin with the prophylactic use of tobramycin-impregnated polymethylmethacrylate beads in compound fractures. Clin Orthop 1988; 237:213–5.
  27. Lerner RK, Esterhai JL, Polomono RC, et al. Psychological, functional, and quality of life assessment of patients with posttraumatic fracture nonunion, chronic refractory osteomyelitis, and lower extremity amputation. Arch Phys Rehab 1991; 72:122–6.
  28. Patzakis MJ, Harvey JP Jr, Ivler D. The role of antibiotics in the management of open fractures.   J Bone Joint Surg Am 1974; 56:532– 41.
  29. Schentag JJ, Lasezkay G, Plant ME, et al. Comparative tissue accumulation of gentamycin and tobramycin in patients. J Antimicrob Chemother 1979; 4(SupplA):23–30.
  30. Seligson D, Popham GJ, Voos K, Henry SL, Faghri M. Antibiotic-leaching from polymethylmethacrylate beads. J Bone Joint Surg Am 1993; 75:714– 20.
  31. Stuart MJ, Morrey BF. Arthrodesis of the diabetic neuropathic ankle joint. Clin Orthop 1990; 253:209– 11.  
  32. Esterhai JL Jr, Sennett B, Gelb H, et al. Treatment of chronic osteomyelitis complicating nonunion and segmental defects of the tibia with open cancellous bone graft, posterolateral bone graft, and soft tissue transfer. Trauma 1990; 30:49–54.
  33. Paul J, Barg A, Horisberger M, Herrera M, Henninger HB, Valderrabano V. Ankle salvage surgery with autologous circular pillar fibula augmentation and intramedullary hindfoot nail. J Foot Ankle Surg. 2014 Sep-Oct; 53(5):601-5.
  34. Klouche S, El-Masri F, Graff W, Mamoudy P. Arthrodesis with internal fixation of the infected ankle. J Foot Ankle Surg. 2011 Jan-Feb; 50(1):25-30.

Acknowledgements: None

Conflicts of Interest: None

Communications Author: Sham Persaud

Level of Evidence: Level IV Therapeutic Study

Early mobilization in bilateral talar fractures

by Mario Cala DPM1, Kristina Barreiro DPM1, Hany Jeffry DPM1.pdflrg

The Foot and Ankle Online Journal 7 (2): 9

Bilateral fractures of the talus can be considered extremely rare. The appropriate treatment suggested by most experts includes permanent anatomical reduction with fixation. Open surgical approaches to the hindfoot can be associated with major complications. Some of these complications include vascular damage, soft tissue and surgical wound-healing problems due to the poor blood supply to the posterior ankle region. To avoid these complications, fluoroscopy assisted closed reduction and percutaneous fixation has been recommended in the treatment of less displaced fractures in some of the literature. This is a case report in which bilateral talar fractures were treated percutaneously resulting in early mobilization.

Key words: talar fracture, early mobilization, external fixation

ISSN 1941-6806
doi: 10.3827/faoj.2014.0702.0009


Address correspondence to: Kristina Barreiro, DPM
Jackson North Medical Center in North Miami, Florida
Email: barreirokristina@gmail.com

1 Jackson North Medical Center in North Miami, Florida


The talus is a very important bone in the lower extremity. The body of the talus is divided into five surfaces: superior, medial, lateral, posterior, and inferior. The anterior surface is attached to the neck. The neck of the talus has four surfaces: superior, medial, lateral, and inferior. The head of the talus is entirely articular, consisting of three articular surfaces; the largest being the talonavicular articulation [1]. It is highly vascularized with three main arteries supplying its blood supply. The anterior tibial artery, posterior tibial artery, and peroneal artery all give branches that supply the talus. Due to its high vascularity, the talus has a high healing rate, if vascularity is not compromised. A positive Hawkins sign is seen in talar fractures 4-8 weeks after the injury at the subcortical bone of the talar dome due to the washing out of the subchondral bone and subsequent osteopenia. This indicates bone remodeling. It is highly predictive of a revitalization of the talar body after a fracture [1].

Talar fractures can vary in site of fracture: from an osteochondral talar dome fracture to a dislocated talar neck fracture to talar body fracture. Each type of fracture has its own outcome in terms of treatment plan due to its mechanism of injury. The mechanism of injury for talar body fractures typically involves hyperdorsiflexion of the foot with impingement of the talar neck against the anterior edge of the tibial plafond [2]. Another mechanism of injury resulting in fracture of the body of the talus is a fall from height, in which an axial compression of the talus between the tibial plafond and the calcaneus results. Fractures of the talar body, while uncommon, present a harsh prognosis due to its greater risk for avascular necrosis than talar neck fractures [1]. Talar dome fractures are caused by two mechanisms of injury: medial lesions were caused by inversion and plantar flexion of the foot with external rotation of the tibia on the talus, while lateral lesions were caused by inversion and dorsiflexion of the foot with internal rotation of the tibia on the talus. Talar neck and head fractures are more common than talar body fractures [3].

Bilateral fractures of the talus can be considered extremely rare. The appropriate treatment suggested by most experts includes permanent anatomical reduction with fixation [3].  One study found a patient with a left talar neck fracture of the lateral process and right talar body fractures with bilateral subtalar and talonavicular dislocations. Treatment was bilateral internal fixation. This case resulted in permanent pain and limitation in movement [3].

The use of plain films and magnetic resonance imaging can help in the diagnosis and staging of these lesions as well as aid in treatment planning. Conservative treatment for nondisplaced talar fractures is common, but displaced fractures require stable fixation and early physical therapy [4]. Classical treatment for talar fractures is open reduction and internal fixation. In the past, crossed k-wires have been used to correct the fractures, as well as cannulated screws, and combinations of k-wires and screws in the talus. Rare cases used mini-plates. Patients who received internal fixation where typically immobilized in a non-weight bearing cast in neutral alignment for a period of twelve weeks [4]. Following the period of non-weight bearing, progressive weight-bearing combined with physiotherapy is usually started.

Arthroscopic treatment may be used in the management of transchondral dome fractures [5]. Following excision of the talar dome fracture fragment, the exposed subchondral bone should be drilled with multiple small drill-holes to promote migration of fibroblasts to the surface for the production of fibrocartilage [6]. Arthroscopically assisted internal fixation of talar body fractures using anterior portals has been noted [5].

Open surgical approaches to the hind foot can be associated with higher complications. Some of these include surgical wound-healing problems due to the poor blood supply to the posterior ankle region. To avoid vascular damage and soft-tissue problems, fluoroscopy assisted closed reduction and percutaneous fixation has been recommended in the treatment of minimally displaced fractures in at least one source [5]. In our case report, in order to minimize complications, we used bilateral mini-rails. The SIDEKICK® Mini Fixator from Wright Medical was utilized in our case. It is indicated to stabilize multiple fracture fragments ranging from open and/or comminuted fractures to infected non-unions, fractures with length discrepancies, fusions and corrective osteotomies of the metacarpal, metatarsal, ulnar, and calcaneal bones [8]. This mini-rail allows early weight-bearing and decreases the risk of avascular necrosis.

Case Report

The patient is a 20-year-old male with no significant past medical history was brought to the emergency department following a motor vehicle accident at 75 MPH. The patient was complaining of ankle pain and swelling, along with left sided chest pain which appeared skeletal.

After a complete series of x-rays and CT scans, he was found to have closed transverse fractures of the body of the talus of right foot (Figures 1, 2, and 5). He also was found to have a minimally displaced fracture of the neck of the talus of left foot that was graded type 1 on Hawkins classification (Figures 3, 4, and 5). No other fractures were seen, and no organ damage was detectable on CT imaging. His physical exam was normal, except for edema and tenderness with limited range of motion of both ankles.

Talar1

Figure 1 Coronal CT view of right rear foot showing comminuted fracture of talar body.

Talar2

Figure 2 Sagittal CT view of right rear foot showing fracture of the talar body.

Talar3

Figure 3 Coronal CT view of left rear foot showing comminuted fracture of talar neck.

Initial management involved pain medication, and immobilization of bilateral ankles utilizing posterior splints with application of modified Jones compressions to control swelling. After medical clearance, surgical intervention was planned for close reduction of the talar fractures of the bilateral feet utilizing mini-rail external fixators.

Talar4

Figure 4 Sagittal CT view of left rear foot showing talar neck fracture.

Talar5

Figure 5 Transverse CT view of bilateral rear foot showing right talar body fracture and left talar neck fracture.

The external fixator was applied to the dorsomedial aspect of the right foot with six half pins (two were inserted into the proximal fragment, two were inserted into the distal fragment, and for more stabilization, one pin into the navicular and one pin into the medial cuneiform bone) (Figure 6). For the left foot, the external fixator was applied to the dorsomedial aspect as well, with two half pins inserted into the proximal fragment, two inserted into the distal fragment, and two pins into the navicular bone to achieve more stabilization (Figure 6). Compression of both tali was achieved by turning the mini-rail bringing the fractured fragments together. Fluoroscopy imaging confirmed good alignment of both tali.

Talar6

Figure 6 Post-operative X-ray of bilateral foot in AP view showing achievement of anatomical alignment of the fractured tali with the assistance of the mini-rail compression and fixation.

The patient received antibiotic before and after the surgery. Postoperatively, we continued with posterior splint and modified Jones compression with non-weight bearing to bilateral foot, assisted with wheelchair. Medications for pain and DVT prophylaxis were prescribed on discharge.

One week after surgery, the patient reported minimal pain to right foot.  He reported walking on his feet disregarding our instructions. Seven weeks after the surgery, the patient reported no pain. X-rays of bilateral feet revealed healing of the talar fractures. He was allowed to bear weight partially assisted with a walker and resume physical activity moderately (Figure 7).

Talar7a Talar7b

Figure 7A & B Clinical pictures of the patient 7 weeks after the surgery date with partial weight bearing and mini-rails bilateral foot.

At nine weeks post operatively, the hardware was removed. Patient tolerated walking without pain and was tolerating physical therapy the week after removing the hardware. X-rays taken one month after removing the mini-rails demonstrated complete resolution of the fracture sites (Figure 8A, B, and C).

Talar8a Talar8b Talar8c

Figure 8A, B, & C Bilateral Foot X-Rays in Lateral and AP views demonstrating complete resolution of bilateral talar fractures post removal of the mini-rail fixator.

Discussion

Because of the young age of the patient, in the hope of achieving earlier weight-bearing with a better chance to be able to return to work, we chose mini-rail as a minimally invasive approach.  Conventional therapies, such as Kirschner wires, would delay early weight-bearing. Our choice of mini-rails also avoided the vascular and wound healing complications that are commonly associated with an open approach to the rear foot. This patient had a chance to heal with no complications, in a shorter time, and was able to ambulate earlier than conventional internal fixation would allow.

References

  1. Morgan A, Kim PS, Christman RA. Radiographic anatomy of the talus. J Am Podiatr Med Assoc. 93 (6): 449-80. – Pubmed
  2. Jimenez AL, Morgan JH. Talar fractures: three case studies. J Am Podiatr Med Assoc. 2001;91 (8): 415-21. – Pubmed
  3. Taraz-jamshidi MH, Shapari O, Shiravani R et-al. Simultaneous bilateral fracture dislocation of the talus: a case report. Trauma Mon. 2013;18 (2): 90-4.  –Pubmed
  4. Ohl X, Harisboure A, Hemery X et-al. Long-term follow-up after surgical treatment of talar fractures: Twenty cases with an average follow-up of 7.5 years. Int Orthop. 2011;35 (1): 93-9. – Pubmed
  5. Ogut T, Seyahi A, Aydingoz O et-al. A two-portal posterior endoscopic approach in the treatment of a complex talus fracture: a case report. J Am Podiatr Med Assoc. 99 (5): 443-6. – Pubmed
  6. Ebraheim NA, Patil V, Owens C et-al. Clinical outcome of fractures of the talar body. Int Orthop. 2008;32 (6): 773-7. – Pubmed
  7. Kilic A, Kabukcuoglu Y, Sokucu S. The treatment of talar body fractures with compression screws: a case series. Cases J. 2009;2 : 7953. – Pubmed
  8. Hollawell, S. SIDEKICK® Mini Fixator: Surgical technique. Wright Medical Technology, Inc. 2013. – link

Calcaneal Intraosseous Lipoma treated with External Fixation: A case report and review of the literature

by James Losito, DPM1, Victor L. Herrera, DPM2, Riquel Gonzalez, DPM3, Thomas Merrill, DPM4

The Foot and Ankle Online Journal 5 (8): 1

A case report is presented of an intraosseous lipoma. Diagnosis was made with the help of Magnetic Resonance Imaging and histopathologic analysis, after which the patient was treated by means of curettage and packing with bone graft substitute. Surgical, histologic features and a staging classification for intraosseous lipoma are presented in this case report. This article also discusses the use of external fixation in a patient with high risk of calcaneal fracture and to promote early weight bearing and early recovery. Although calcaneal intraosseous lipoma accounts for a small portion of cases in the huge differential diagnosis chart for foot pain such as plantar fasciitis, retrocalcaneal bursitis, gout and stress fracture, it should be kept in mind as a possible diagnosis in cases of unresolved pain to the heel.

Key Words: Intraosseous lipoma, external fixation, heel pain, bone tumor.

Accepted: July, 2012

Published: August, 2012

ISSN 1941-6806
doi: 10.3827/faoj.2012.0508.0001


Intraosseous lipomas are rare benign bone tumors. This benign neoplasm has been reported to occur in the calcaneus as well as the proximal femur [1,6,7]. In the past, the relative absence of symptoms and radiographic similarity to a bone cyst has accounted for under diagnosis of intraosseous lipoma [2,3]. Intraosseous lipomas are derived from mature lipocytes mostly seen at the metaphysis of the long bones in men [5,6]. Foot and heel pain are the common symptoms of calcaneal intraosseous lipoma [5].

Non-surgical options such as NSAIDs, cold compression, use of non-weight bearing devices such as cane, use of silicone sole plate and preventive measures for pathological fractures are the most commonly used treatment modalities for this condition. Surgery is indicated in the presence of pain resistant to conservative treatment methods, impending or pathological fractures and when a histopathological differential diagnosis is required for aneurismal bone cyst, giant cell tumor, pseudo cyst formation or unicameral bone cyst. Although surgical treatment with curettage and autogenous bone grafting has been reported as a treatment choice, only small case series have been reported thus far [10]. In this study, we present one calcaneal intraosseous lipoma in a patient treated with curettage, autogenous bone grafting and Ilizarov external fixator.

Case Report

A 23 year-old male presents with the complaint of a dull, aching pain in his right heel of 2 years of duration. The pain was noted to increase after strenuous walking, long time of standing or other rigorous activities involving the right foot. The pain has been increasing steadily over the 3-4 months period. Patient stated the pain is persistent and worse at the end of the day. The pain is ranked (7 out of 10) on pain scale with 0 no pain, and 10 severe pain. There is associated swelling observed of the right foot.

 

Figures 1A and 1B Lateral plain x-rays (A) and antero-posterior views (B) with well-circumscribed bone lesion.

On gross examination, the patient walked with an antalgic gait. There is a small soft tissue palpable fluent mass on the right medial arch below the medial malleolus compatible with a possible superficial soft tissue lipoma. No scars, sinuses or venous prominences overlying the affected area, and the right ankle and subtalar joint motions were normal.

There is pain on palpation to the right heel and ankle. There is no past medical history that would increase the likelihood of bone infarction, such as corticosteroid use, infection, previous irradiation, lipid storage disease, collagen-vascular disease, or lympho-proliferative disorder.

Diagnosis

Plain radiographs revealed the presence of a well-circumscribed radio-mixed lesion with a thin sclerotic rim, interspersed with trabeculations in the antero-inferior portion of the left calcaneus underlying the subtalar joint. (Figs.1A and 1B) A preoperative magnetic resonance imaging (MRI) scan of the Right foot reveals the presence of a 2.3 x 2.0 cm circumscribed mass to the neck and body of the calcaneus. Predominant fat signal is seen on all pulse sequences. There is an eccentric component of fluid signal within the lateral aspect of the mass (Figs. 2A, 2B, 2C and 2D). The appearance of the mass is compatible with an intraosseous lipoma. There is prominent fatty tissue seen in the plantar, medial aspect of the right hindfoot most likely represents a prominent lobule of subcutaneous fat. It is at this point that the surgical option was discuses with the patient and he agreed to undergo surgical approach of his condition.

   

Figures 2A, 2B, 2C and 2D T1/T2 sagittal images shows fluid signal within the lateral aspect of the mass. (A and B) T1/T2 coronal images with well demarcated mass in the calcaneus. (C and D)

Surgical Approach

Based on the clinical and diagnostic image findings, intraosseous lipoma is diagnosed and operative decompression of the cyst is subsequently undertaken. Prior to the operation, the lesion is localized fluoroscopically and its localization is marked on the skin. Under tourniquet control, a straight lateral skin incision is performed over the lesion and the periosteum is incised longitudinally. The lesion and a portion of the adjacent normal tissue were exposed at one end of the lesion using a 1cm×1 cm rectangular cortical window. The cortex overlying the cyst is exposed on the inferior and lateral aspects. Using an oscillating saw and osteotome the cortex is opened and the lesion is totally curetted out with angled curettes through the cortical window.

As the cyst is decorticated, a greasy-yellow intraosseous lipoma is identified and evacuated from the osseous cavity. The soft tissue contents of the intraosseous cyst were removed along with the greasy fluid and the entire specimen is sent for histopathologic diagnosis. The cavity of the calcaneus is lavaged with normal saline before cancellous allograft bone is used to pack the cavity. After filling the cavity, the wound is closed in anatomic layers and a sterile dressing applied, followed by application of an Ilizarov ring external fixator for the initial postoperative period to allow for weight-bearing ambulation (Figs. 3A and 3B). Postoperative radiographs show the orientation of the external fixator to allow for early amputation after surgery (Figs. 4A and 4B).

 

Figures 3A and 3B Ring External Fixation system applied after the excised the bone tumor.

 

Figures 4A and 4B Lateral and A-P views of Post-Op X-Rays evaluation.

Subsequent histopathologic analysis reveals fragments of bone which include a few fragments of necrotic bone and fibroadipose tissue which shows foci of fat necrosis and necrosis of other soft tissue-types. The morphology suggests a possible fracture site or tendon avulsion. There is no evidence of neoplasm. These findings are consistent with the diagnosis of intraosseous lipoma. The patient’s heel pain subsided almost immediately after the operation, with the exception of surgical wound pain, which subsided in normal fashion.

A postoperative magnetic resonance imaging (MRI) scan of the right] foot is done 3 months after surgery once the fixator is removed. This reveals the resection of the previously described intra-osseous fatty mass in the neck and body of the calcaneus. Intermediate signal intensity tissue now fills this region of the calcaneus. There is no calcaneal fracture identified (Figs. 5A, 5B, 5C and 5D).

   

Figures 5A, 5B, 5C and 5D MRI shows T1/T2 sagittal views. (A and B) T1/T2 axial views of 12weeks MRI follow up evaluation shows bone graft uptake and reduced size of the bone cavity without fluid signal. (C and D)

Two weeks following suture removal, the patient is mobilized with instructions for partial weight bearing in the following 3 weeks, followed thereafter by weight bearing as tolerated. Clinical and radiological examinations are performed on the first postoperative day, at 6weeks, at 12weeks and every other month thereafter, until there is radiological confirmation of graft consolidation (Figs. 6A, 6B, 6C and 6D).

   

Figures 6A, 6B, 6C and 6D Clinical examinations at 12weeks post-op, without External Fixation. There is now good and adequate ankle range of motion.

Discussion

Milgram’s classification system is used for staging the lesions: In stage 1, the lesion is a solid lipoma composed of viable fat cells; in stage 2, part of the lesion is necrotized, forming focal calcification; and in stage 3, most of the tumor tissue has died, with variable degrees of cyst formation, calcification, and reactive new bone formation [19].

Histopathologic analysis of our (specimen) reveals fragments of bone which include a few fragments of necrotic bone and fibroadipose tissue showing foci of fat necrosis and necrosis of other soft tissue-types. The morphology suggests a possible fracture site or tendon avulsion. There is no evidence of neoplasm. These findings were consistent with the diagnosis of stage 2 intraosseous l intraosseous.

The need for surgical treatment is controversial. Curettage with bone grafting is the treatment of choice when surgical intervention is needed. Most lipomas, however, can be managed conservatively. Some surgeons feel that in asymptomatic cases with no signs of an impending pathologic fracture or suspicion of malignancy that a non-operative treatment with clinical and radiological follow-up is indicated. Malignant transformation is rare. While some surgeons think that biopsy is unnecessary because radiological features are characteristic, others believe that the lesion must be diagnosed histologically. However, reports stating that biopsy is required usually predate the common and efficient use of MRI, when an accurate radiological diagnosis was almost impossible.

We believe that pain alone is not an indication for surgical intervention or any other invasive treatment, including biopsy. The cause of pain in the patient with intraosseous lipomas is unclear, but it may be mechanical due to expansile remodeling of bone. It may be related to the ischemic changes that frequently accompany these lesions. It is also possible that the pain is referable from nearby joint disease and that the an intraosseous lesion is incidentally discovered. It is reported that symptoms may recur after surgical treatment or resolve spontaneously on conservative treatment, thus suggesting that many intraosseous lipomas are incidental findings and that patients may have another, unidentified cause of symptoms. Microtrabecular fracture in areas of weakened bone following episodes of minor trauma may be one cause of pain. Areas of diffuse increased signal were observed on MRI within the lipoma in some series, which may represent a stress response.

Asymptomatic intraosseous lipomas of the calcaneus should not require surgical intervention, since the tumor always occurred in the region of Ward’s triangle, which is a non weight-bearing region. In fact, in healthy individuals it is a region with bone paucity. A pathological fracture seems to be unlikely and has not been previously reported in the calcaneus.

Small cysts that are not located in the pressure-bearing trabecular area of the calcaneus are usually asymptomatic and can be treated conservatively. A “critical-size cyst” has been defined as an intraosseous lipoma extending the full breadth of the calcaneus laterally to medially in the coronal plane, and occupying at least 30% of the length of the calcaneus anteroposterior. Since the presence of a pathological fracture through a calcaneal cyst makes the operative procedure more complex and healing less predictable, the authors believe that large symptomatic calcaneal cysts should be managed surgically to reduce morbidity.

The decision to operate on a calcaneal cyst should be based on its size and location, the provisional diagnosis, associated symptoms, and the activity level and health of the patient. Although intraosseous lipoma is a benign lesion, Milgram [19] described four cases of intraosseous lipoma that underwent malignant transformation. Liposarcoma and malignant fibrous histiocytoma have also been found adjacent to benign lipomas.

Treatment of intraosseous lipomas is still controversial. Hirata, et al.[30], suggested that surgical treatment is not necessary owing to the potential for spontaneous regression and very low rate of malignant transformation. However, according to Weinfeld, et al.[21], curettage and grafting is the best choice of treatment. Schneider stated that the need for surgical treatment relies on the risk of malignant transformation [23]. Bertram reported a 33% rate of accidental diagnosis among 54 patients and surgery was only required when the patient was clinically symptomatic [24].

Gonzalez’s conclusion was similar to Bertram and stated that the majority of calcaneal intraosseous lipomas are seen in Ward’s triangle [10]. According to Mollin, et al.[28], curettage and grafting is a good choice for permanent treatment and can be performed if the patient is symptomatic. In the present case report, we operated on a symptomatic patient. He was resistant to conservative treatment for the previous 3 to 6 months. He underwent surgery due to the pain, incapacity to perform any sport activity and a suspicion for risk of pathological fracture due to his athletic ability.

Our patient started weight bearing just after surgery with an external fixator. He recovered with full benefit after nine months, and increased his sport activities without any complaint. Since intraosseous lipoma is an uncommon bone tumor, there is a need to familiarize physicians with the radiographic and MRI features of this lesion for the correct diagnosis and treatment.

References

1.  Kapukaya A, Subasi M, Dabak N, Ozkul E. Osseous lipoma: eleven new cases and review of the literature. Acta Orthop Belg 2006 72: 603-614. [PubMed]
2.  Radl R, Leithner A, Machacek F, Cetin E, Koehler W, Koppany B, Dominkus M, Windhager R. Intraosseous lipoma: retrospective analysis of 29 patients. Int Orthop 2004 28: 374-378. [PubMed]
3.  Campbell RS, Grainger AJ, Mangham DC, Beggs I, Teh J, Davies AM. Intraosseous lipoma: report of 35 new cases and a review of the literature. Skeletal Radiol 2003 32: 209-222. [PubMed]
4.  Reig-Boix V, Guinot-Tormo J, Risent-Martinez F, Aparisi-Rodriguez F, Ferrer-Jimenez R. Computed tomography of intraosseous lipoma of os calcis. Clin Orthop Relat Res 1987 (221): 286-291. [PubMed]
5.  Yildiz HY, Altinok D, Saglik Y. Bilateral calcaneal intraosseous lipoma: a case report. Foot Ankle Int 2002 23: 60-63.  [PubMed]
6.  Buckley SL, Burkus JK. Intraosseous lipoma of the ilium. A case report. Clin Orthop Relat Res 1988 (228): 297-301. [PubMed]
7.  Arslan G, Karaali K, Cubuk M, Senol U, Lüleci E. Intraosseous lipoma of the frontal bone. A case report. Acta Radiol  2000 41: 320-321. [PubMed]
8.  Kapukaya A, Subasi M, Dabak N, Ozkul E. Osseous lipoma: eleven new cases and review of the literature. Acta Orthop Belg 2006 72: 603-614. [PubMed]
9.  Chow LT, Lee KC. Intraosseous lipoma. A clinicopathologic study of nine cases. Am J Surg Pathol 1992 16: 401-410. [PubMed]
10.  Gonzalez JV, Stuck RM, Streit N. Intraosseous lipoma of the calcaneus: a clinicopathologic study of three cases. J Foot Ankle Surg 1997 36: 306-310. [PubMed]
11.  Propeck T, Bullard MA, Lin J, Doi K, Martel W. Radiologic-pathologic correlation of intraosseous lipomas. AJR Am J Roentgenol2000 175: 673-678.  [PubMed]
12.  Kamekura S, Nakamura K, Oda H, Inokuchi K, Iijima T, Ishida T. Involuted intraosseous lipoma of the sacrum showing high signal intensity on T1-weighted magnetic resonance imaging (MRI). J Orthop Sci 2001 6:183-186. [PubMed]
13.  Levin MF, Vellet AD, Munk PL, McLean CA. Intraosseous lipoma of the distal femur: MRI appearance. Skeletal Radiol 1996 25: 82-84.  [PubMed]
14.  Blacksin MF, Ende N, Benevenia J. Magnetic resonance imaging of intraosseous lipomas: a radiologic-pathologic correlation. Skeletal Radiol1995 24: 37-41. [PubMed]
15.  Rosenblatt EM, Mollin J, Abdelwahab IF. Bilateral calcaneal intraosseous lipomas: a case report. Mt Sinai J Med 1990
57: 174-176. [PubMed]
16.  Ketyer S, Brownstein S, Cholankeril  J.  CT diagnosis of intraosseous lipoma of the calcaneus. J Comput Assist Tomogr 1983 7: 546-547. [PubMed]
17.  Kozlowski K, Welshman R. What is it? Intraosseous lipoma in a 13-year-old boy. Br J Radiol 1991 64: 855-856. [PubMed]
18.  Lagier R. Case report 128. Skeletal Radiol 1980 5: 267-269, 1980. [PubMed]
19.  Milgram JW. Intraosseous lipomas. A clinicopathologic study of 66 cases. Clin Orthop 1988 231: 277-230. [PubMed]
20.  Poussa M, Holmstrom T. Intraosseous lipoma of the calcaneus. Report of a case and a short review of the literature.  Acta Orthop Scand 1976 47: 570-574. [PubMed]
21.  Weinfeld GD, Yu GV, Good JJ. Intraosseous lipoma of the calcaneus: a review and report of four cases. J Foot Ankle Surg 2002 41: 398-411.  [PubMed]
22.  Schneider O, Mischo J, Puschel W. Intraosseous lipoma of the calcaneus. Chirurg 1994 65: 74-76. [PubMed]
23.  Bertram C, Popken F and Rutt J. Intraosseous lipoma of the calcaneus. Congen Arch Surg 2001 386: 313-317. [PubMed]
24.  Langenbecks, Tejero A, Arenas AJ and Sola R. Bilateral intraosseous lipoma of the calcaneus. A case report. Acta Orthop Belg 1999 65: 525-527.  [PubMed]
25.  Rosenblatt EM, Mollin J and Abdelwahab IF. Bilateral calcaneal intraosseous lipomas: a case report. Mt Sinai J Med 1990 57: 174-176.  [PubMed]
26.  Ramos A, Castello J, Sartoris DJ, Greenway GD, Resnick D, Haghighi P. Osseous  lipoma: CT appearance. Radiology  1985 157: 615-619. [PubMed]
27.  Bruni L. The “cockade” image: a diagnostic sign of calcaneum intraosseous lipoma. Rays 1986 11: 51-54.  [PubMed]
28.  Reig-Boix V, Guinot-Tormo J, Risent-Martinez F, Aparisi-Rodriguez F, Ferrer-Jimenez R. Computed tomography  of intraosseous lipoma of os calcis. Clin Orthop 1987 221:286-291. [PubMed]
29.  Hirata M, Kusuzaki K and Hirasawa Y. Eleven cases of intraosseous lipoma of the calcaneus. Anticancer Res 2001 21:  4099-4103. [PubMed]


Address correspondence to: Victor Herrera, DPM email: herreragioco@bellsouth.net

1Diplomate, American Board of Podiatric Surgery, American Academy Podiatric Sports Medicine.
2Senior Resident at Barry University/ Mercy Hospital, Miami, Florida
3Resident at Barry University/ Mercy Hospital, Miami, Florida
4Diplomate, American Board of Podiatric Surgery.

© The Foot and Ankle Online Journal, 2012

Arthrodiatasis in the Treatment of Ankle Arthritis: A Case Series

by Edgardo Rodriguez, DPM1, Byron Hutchinson, DPM2, Craig Clifford, DPM3, Kevin McCann, DPM4

The Foot and Ankle Online Journal 5 (7): 2

Ankle arthritis that has failed conservative treatment warrants a more aggressive approach. Most treatments for ankle arthritis are primarily joint destructive, with a high probability for long term negative sequelae. The option to attempt surgical treatment with a less invasive procedure is appealing for both the surgeon and the patient. The purpose of this study is to demonstrate the efficacy of ankle arthrodiatasis with the use of external fixation as an alternative treatment for ankle arthritis. Eighty-two patients were evaluated preoperatively and postoperatively for pain, function, and complications with the Maryland foot score. Twenty patients (24%) experienced excellent results, forty-five (55%) good results, twelve patients (12%) had fair results, and five patients (6%) had poor results. Of the five poor results, four patients underwent an ankle replacement and one patient underwent ankle arthrodesis. The authors consider the use of ankle distraction with ankle arthroplasty as a viable alternative to previously accepted treatments for severe ankle arthritis. The hallmark benefit of this procedure is its joint sparing properties. Decreased soft tissue dissection associated with the use of external fixation makes this less invasive treatment available to a wide range of patients. Ankle arthrodiatasis is a viable treatment option for the treatment of advanced ankle arthritis.

Key Words: Arthrodiatasis, Ankle Distraction, Distraction Arthroplasty, External Fixation, Ankle Arthritis

Accepted: June, 2012

Published: July, 2012

ISSN 1941-6806
doi: 10.3827/faoj.2012.0507.0002


Articular damage to the ankle joint has a broad range of etiologic processes including traumatic arthritis, primary degenerative osteoarthritis, neuropathic arthropathy, inflammatory arthritis, and infectious arthritis.

Because of pain when ambulating or inability to bear weight, these ankle arthritides can be exceedingly debilitating. The most common causes of degenerative changes in the ankle joint are previous trauma to the joint and abnormal ankle mechanics [1]. Unlike the larger, more proximal joints of the lower extremity, primary idiopathic osteoarthritis is not the leading cause of damage to ankle joint articular cartilage.

It has been previously shown that up to 70% of symptomatic ankle arthritis is associated with past rotational ankle fractures or other previous trauma, with only 7% manifesting as primary (idiopathic) osteoarthritis [2].

Ankle arthritis may be evaluated both clinically and radiographically. On examination, patients may present with painful and limited joint motion, tenderness, variable degrees of inflammation, joint crepitus, and osseous deformity. Evidence of joint space narrowing, osteophyte formation, subchondral cystic lesions, and osseous erosions are often visible radiographically (Figs 1A and 1B). Early symptoms of ankle arthritis can be treated successfully with several conservative therapies. Pharmacologic agents, such as non-steroidal anti-inflammatory drugs (NSAIDs), intra-articular steroidal injections, and visco-supplementation with sodium hyaluronate injection may be beneficial in reducing symptoms [3]. Lifestyle changes, which include weight reduction and the use of bracing devices such as Ankle-Foot Orthoses have shown to decrease symptoms as well. When conservative measures fail and symptoms persist a more aggressive approach is warranted, which includes operative intervention.

 

Figure 1A and 1B Preoperative anterior-posterior radiograph of arthritic ankle joint (A). Preoperative lateral radiograph of arthritic ankle joint showing extensive tibial hypertrophy (B).

To date, ankle arthrodesis for the treatment of end-stage ankle arthritis has yielded good short- and intermediate-term results. Several reports [4,5,6] have suggested that primary arthrodesis relieves symptoms in approximately 80% of treated individuals by providing a stable, plantigrade, and ideally a painless foot. Yet even when performed appropriately, this procedure has its inherent complications, including malalignment, abnormal biomechanics, painful retained hardware, and most importantly premature arthritis of adjacent joints. With advances in joint replacement technology, new generation ankle prostheses are designed to more accurately mimic the true anatomy and biomechanics of the ankle. Unlike early counterparts, these second generation implants have exhibited promising results [7]. Ankle joint replacement however, is a difficult procedure to master, and fraught with the possibility of long term complications and eventual implant failure. With a high technical learning curve, these procedures should only be performed by skilled surgeons with adequate procedural volume. Salvage procedures after failed ankle replacement may include revision, arthrodesis, and even amputation [8].

The term “arthrodiatasis” was coined in 1979 in Verona, Italy. Its origin stems from the Greek words for “joint” (arthros), “through” (dia), and “to stretch out” (tasis) [9]. In 1975, Volkov and Oganesian first reported the use of joint distraction in the knee and elbow [10]. Only recently has literature started to surface which studies distraction for the treatment of arthritic ankles. The first documented case of ankle arthrodiatasis was published in 1978 [9,11], but the majority of work with ankle distraction began in the 1990’s. Van Valburg et al. reported on joint distraction utilizing an Ilizarov apparatus in 11 patients, resulting in increased joint space as well as decreased pain and improved mobility for a mean of two years. Van Roermund, van Valburg, and their team in the Netherlands have led much of the research on ankle distraction [12-18].

Ankle arthrodesis and implant arthroplasty are both joint destructive procedures with a high possibility for long term negative sequellae [19].

Ankle arthrodiatasis with the use of an external fixator carries the primary benefit of being non-destructive to the ankle joint, which retains for the younger patient the possibility of later implant arthroplasty or arthrodesis. The purpose of this study is to demonstrate the efficacy of ankle arthrodiatasis with the use of external fixation as an alternative treatment for ankle arthritis.

Methods

Eighty-two distractions were performed in 82 non-consecutive patients from 1998 to 2010, with a mean age of 49 years of age, and follow-up ranging from one to 12 years. Of these, 45 were male and 37 female. Thirty-seven (45%) of patients had a deformity correction (i.e. midfoot, calcaneal, or supramalleolar osteotomy) and twenty-three (28%) underwent an Achilles tendon lengthening. Seventy-Eight of 82 patients (95%) underwent an open ankle arthrotomy at the time of frame application to remove any impinging anterior osteophytes and improve ankle joint range of motion (Figs. 2A and 2B). All patients had arthritis of the ankle joint secondary to trauma and were recommended for an arthrodesis by other treating physicians after failing conservative treatment. All patients had either slight or marked decrease in range of motion at the ankle as compared to accepted normal values with none of the ankles being graded as ankylosed.

 

Figure 2A and 2B Open arthrotomy with visible osteophyte formation (A). Open arthrotomy following resection of anterior tibia (B).

All patients were assessed by one of the two senior authors (ER or BH) both preoperatively and postoperatively using the Maryland foot score [20].

Patient Preparation and Additional Procedures

Operative procedures were performed in the supine position under general anesthesia with a thigh tourniquet. If indicated, adjunctive procedures were performed at the time of frame application. A percutaneous tendoAchilles lengthening was performed to correct ankle joint equinus. An osteotomy was performed in the calcaneus to correct for a varus deformity, or in the tibia to correct for a procurvatum deformity if indicated.

Aggressive ankle debridement at the time of ankle distraction was performed from either an open anterior medial approach or arthroscopically to increase intraoperative range of motion and remove osteophytic blocks and soft tissue impingement. An accessory anterior lateral incision was necessary as well in some cases. After performing the necessary ancillary procedures, incision sites were closed appropriately via the surgeon’s preference, and the pneumatic thigh tourniquet was deflated.

Frame Preparation and Wire Placement

The authors utilized a multi-planar ring external fixation system for arthrodiatasis. Three main objectives of frame design for ankle distraction are: providing stability, allowing sufficient room for soft tissue clearance, and permitting weight bearing as soon as possible. The frame was pre-built in order to minimize intraoperative anesthesia time. It was generally comprised of two proximal rings, which were attached to the tibia, and a distal foot plate or one-third ring, which was attached to the foot. The two tibial rings and the distal foot plate or one-third ring were separated by threaded rods, which varied in length depending on patient size. Three or four threaded rods separated each tier of the frame. Ring size was chosen to allow two finger-breadths between the ring and the leg at any point. Rings that are too large provide less rigid fixation, but it is essential that there is enough space to allow for postoperative swelling [20].

The proximal ring was positioned perpendicular to the axis of the tibial shaft and the limb was centered within the ring. The ankle joint was placed at 90 degrees, with the foot in a neutral position. The distal one-third ring or foot plate was placed parallel to, but not distal to the plantar surface of the foot, so that patients were able to bear weight directly on the plantar foot with minimal interference from the distal frame. Smooth wires were utilized to attach the frame to the extremity, consisting of two crossing wires through the calcaneus distal to the neurovascular bundle, and two crossing wires at each tibial ring level. A third wire was placed at each tibial ring if the patient weighed more than 200 pounds. All wires were secured and tensioned to their corresponding ring.

Distraction Technique

Distraction was accomplished by tightening nuts along threaded rods or with telescoping rods connecting the foot plate to the distal tibial ring for six millimeters of acute distraction. Fluoroscopic imaging was utilized to verify distraction length. Vascular supply and small vessel integrity were evaluated after distraction via palpation of pedal pulses and capillary refill time. In postoperative recovery if the patient was unable to tolerate the distraction, the amount of distraction was reduced until the patient was comfortable.

Gradual distraction was accomplished at a rate of one millimeter per day in four separate daily adjustments until the desired amount of distraction was obtained.

Postoperative Management

The space between the skin and the fixator rings was packed with a bulky bolster dressing in an effort to control postoperative edema. This is particularly important in the ankle and heel region where swelling is most severe after this type of procedure. Postoperative dressings remained in place for three to seven days. Distraction was confirmed radiographically at the first postoperative visit. (Figs. 3A and 3B) At time of dressing change, pin sites were cleansed with isopropyl alcohol. After approximately two weeks postoperatively, patients began daily self-care of the pin sites with the use of isopropyl alcohol. Sutures and staples remained intact until the frame was removed. Patients were allowed to begin wetting the frame in the shower after approximately two weeks, when the pin sites were dry and stable.

 

Figure 3A and 3B Postoperative anterior-posterior radiograph depicting arthrodiatasis (A). Postoperative lateral radiograph depicting arthrodiatasis (B).

Patients began partial weightbearing as soon as possible after the procedure. Under most circumstances, the patients were encouraged to begin touchdown weight bearing on the first postoperative day as tolerated. Physical therapy was started while patients were in the hospital to instruct the patient with gait utilizing an assistive device, usually a walker.

The ring fixator was removed under general anesthesia after 10 to 12 weeks if a corresponding osteotomy had been performed, and after approximately six weeks if no osteotomy was performed. In most cases, the ankle joint was arthroscopically debrided, and lateral ligamentous laxity of the joint was assessed and repaired if necessary. Radiographs were taken at the time of frame removal (Fig. 4). The patients began weight bearing immediately after the frame was removed with a fracture walker. Patients were able to bear full weight with the use of the fracture walker, but often required a gait assistive device such as a cane or crutch for a period ranging from several weeks to about two months after the frame was removed. Transition to regular shoe gear occurred at two to four weeks, depending upon whether or not ligamentous repair was necessary.

Figure 4 Same patient as in Figure 1, following arthrodiatasis and frame removal.

Results

Patients were evaluated preoperatively for pain and function, and at most recent follow-up for pain, functional outcome, and complications. A retrospective chart review was performed, and patients were assigned to outcomes groups of excellent, good, fair, or poor based on the modified Maryland foot score. All patients rated their cosmetic results as acceptable postoperatively.

Twenty patients (24%) had an excellent outcome. These patients reported minimal pain and were able to walk unlimited distances. These patients did not experience weakness, and did not require a supportive device.

They related the ability to ambulate in any shoes, or shoes with only mild concessions. They also related the ability to ambulate on any terrain and climb stairs normally without difficulty. The 45 patients (55%) with good results experienced mild to moderate pain and were only slightly limited in walking distance. They experienced mild weakness not requiring a supportive device. They related the ability to ambulate in shoes with minor concessions or with orthotics. These patients experienced difficulty ambulating on rocks and hills. Several also reported requiring a banister or other method of assistance when climbing stairs.

Twelve patients (15%) had fair results, and usually related moderate pain and were slightly to moderately limited in walking distance. They typically experienced mild to moderate weakness with only one patient requiring a supportive device. They were able to ambulate in shoes with orthotics, experienced difficulty ambulating on rocks and hills, and reported requiring some method of assistance when climbing stairs. Finally, five of the patients (6%) had a poor outcome following distraction. Four of these patients ultimately required an ankle replacement and one required ankle arthrodesis.

The most common complication encountered was pin site irritation or infection in 12 patients (15%), which is commonly seen with external fixation. Clinical signs of local infection were treated successfully with use of aggressive cleansing of pin sites and with oral antibiotics if necessary. One patient (1.2%) developed acute osteomyelitis which was resolved with intravenous antibiotics. One patient (1.2%) was removed from the multiplanar ring fixator in four weeks secondary to psychological intolerance to the device. Ligamentous laxity after distraction occurred with two patients (2.4%), and was corrected with either bracing or lateral ligament repair. One patient (1.2%) developed a deep vein thrombosis, leading to a pulmonary embolism. This was treated and completely resolved via anticoagulant therapy. One patient (1.2%) developed Charcot neuroarthropathy of the midfoot. Fortunately, this development was identified and treated promptly, and did not change the patient’s overall functional outcome.

Discussion

Advanced arthritis of the ankle joint can be one of the most difficult and debilitating pathologies treated by the foot and ankle specialist. Although no single treatment is appropriate for every patient, the authors consider the use of ankle distraction with arthroplasty as a viable alternative to previously accepted treatments. Decreased soft tissue dissection associated with the use of the external fixation device as compared to more aggressive procedures make this treatment available to a wide range of patients. It has been advocated [9] for patients under the age of 50, to delay arthrodesis procedures by five to ten years and opt for other operative procedures. Stress placed on adjacent joints following ankle arthrodesis often leads to arthritic changes in these joints as well. It follows therefore that arthrodesis of the ankle joint should be avoided in younger patients. The non-destructive nature of arthrodiatasis creates an additional option to delay arthrodesis provided the patients are proper candidates for the procedure and understand the possible need for a more aggressive procedure later on in life.

Few studies have been published on the use of ankle distraction as treatment for severe ankle arthritis. In 1995, van Valburg et al. [17] reported on joint distraction utilizing an Ilizarov apparatus in 11 patients with posttraumatic arthritis. Improvement in pain and mobility were noted. Ankle range of motion increased by 55% and joint space widening was seen in 50% of the patients radiographically. Van Valburg, et al,. [15] published a two year prospective follow up in 17 patients, indicating that 66% continued to have symptomatic relief. In 1998, van Roermund, et al,. [16] presented three cases of joint distraction for arthritis. The joints distracted in this case study were the interphalangeal joint of the thumb, the patellofemoral joint, and the ankle joint. Van Roermund et al. [14] later implied that joint distraction in the case of severe ankle osteoarthritis may be a treatment of choice. In 2002, Marijnissen, et al,. [13] reported significantly better results with ankle distraction than with debridement alone. Marijnissen, et al,. [12] then advocated the use of ankle joint distraction as the treatment of choice in patients of a relatively young age with severe ankle arthritis.

In a study reporting the effects of joint distraction in a canine model, van Valburg reported that in the arthritic canine knee joint distraction produced a return to control levels of abnormal cartilage proteoglycan as well as a decrease in local inflammation, suggesting a change in cartilage metabolism [18]. Chiodo and McGarvey advocated further study of ankle distraction due to its minimally invasive nature, combined with the fact that it is not joint-destructive [23]. Even if joint distraction provides only temporary relief and clinical results slowly deteriorate over time, more definitive and committed procedures can potentially be postponed for a considerable period of time. Ploegmakers, et al,. reported six of 22 patients (73%) treated with ankle distraction showed significant improvement in symptoms at seven years postoperatively [24]. Most recently, Paley and Lamm have performed 20 ankle joint distractions using a hinged external fixator, allowing for range of motion within the ankle joint [9]. Eighteen of the distracted joints were rated good or excellent, with a follow up ranging from two to 17 years.

In theory, distraction of the ankle joint allows for maintenance of intermittent intra-articular fluid pressure, thereby promoting cartilage reparative processes [10]. Damage to the ankle joint is further diminished by offloading contact between the joint surfaces. Subchondral sclerosis is reduced during distraction, which decreases the mechanical stresses on the cartilage during loading of the joint and allows for greater absorption of stresses during ambulation [9,13,14,23].

As noted previously, there is a much lower incidence of primary osteoarthritis in the ankle compared to the knee. Studies performed by Cole, et al,. [25] compared the cartilage between the human talocrural and tibiofemoral joint. They were able to demonstrate that the ankle joint had better reparative processes compared to the knee joint. The biochemical composition of the ankle joint has a more dense extracellular matrix, which resists loading and is less prone to damage [25,26,27]. This may cause a shift in the distracted joint, favoring cartilage synthesis rather than degradation.

The ability of ankle cartilage to repair itself along with the documented clinical benefits of ankle distraction has caused increased interest in this particular joint sparing operative procedure.

As is the case with almost all operative procedures, proper patient selection is a key component to increasing the chance for a successful outcome. Radiographic criteria, concurrent lower extremity deformities, the age and overall health of the patient, and the patient’s motivation and willingness to comply with instructions are all key factors to consider when deciding whether ankle distraction is appropriate for a patient.

Before considering patients for ankle distraction, the surgeon must evaluate and address any adjacent lower extremity deformities. Additional procedures may be indicated to create a stable, plantigrade foot, which is necessary for a successful outcome after ankle distraction. The surgeon must also be prepared to address any talar dome lesions that may be present, either arthroscopically or with open arthrotomy.

Standard weight bearing radiographs of the foot and ankle provide sufficient preoperative imaging for most distraction procedures. Hindfoot alignment views or other imaging modalities may be necessary to evaluate more complex deformities. Common findings among post-traumatic arthritic joints, which are most prevalent with ankle arthritis, include joint space narrowing, subchondral sclerosis, osseous erosions, and osteophyte formation. Relative radiographic contraindications to performing an ankle distraction include flattening of the talar dome, presence of greater than five degrees of valgus or varus deformity of the hindfoot, midtarsal joint degenerative joint disease, severe forefoot abnormalities, and severe equinus deformity.

Generally, ankle arthrodiatasis is indicated for younger or more active patients experiencing pain, instability, and deformity but seeking alternatives to ankle arthrodesis and replacement. Patient age is less important than the individual’s overall health, compliance, motivation, and ability to tolerate rigorous post-distraction rehabilitation and physical therapy.

Distraction procedures have been shown to produce slow, progressive improvement over a period of many months with most patients noticing the greatest amount of functional and symptomatic improvement approximately one year after surgery [14].

Contraindications to performing this procedure include medical co-morbidities that preclude the patient from undergoing an elective operative procedure, severe osteoporosis, infection, non- reconstructable malalignment of the lower extremity, severe peripheral vascular disease and neuroarthropathy. Psychosocial issues must also be addressed prior to frame placement.

The most common complication associated with external fixation procedures is pin-tract infections [28]. Fortunately, these infections are usually superficial and localized, making them manageable to treat. The infections often begin as a cellulitis secondary to Staphylococcus aureus, and they respond quickly to oral antibiotics [29]. If the infection involves deeper tissues and/or bone, the patient may require intravenous antibiotic therapy, with or without wire removal [31]. Pin-site care opinions differ, but a study by Davies, et al., showed that pin-sites and wires managed with the technique used by the Russian Ilizarov Scientific Centre for Restorative Traumatology and Orthopaedics were less likely to develop pin tract infections [29]. This technique focuses on avoiding thermal injury and local formation of hematoma during surgery and utilizing alcoholic antiseptic and occlusive pressure dressings postoperatively.

Neurovascular injury during pin placement can generally be avoided with proper planning and a firm grasp of the cross sectional anatomy in each region of wire placement. Posterior tibial nerve traction injuries and tarsal tunnel syndrome are also possible complications associated with ankle distraction procedures [30]. Gradual distraction of the ankle can sometimes prevent this complication. Another option for prevention is to perform a prophylactic tarsal tunnel release at the time of ankle distraction. The posterior tibial nerve is at risk for traction injuries during acute or gradual correction of hindfoot deformities.

The addition of this procedure has been shown to relieve a considerable amount of postoperative nerve traction and tarsal tunnel symptoms [31,32].

Venous thrombosis is always a risk after procedures in which external fixators are applied, and the surgeon should use prophylactic measures to prevent this. Low molecular weight heparin (LMWH) is started approximately 12 to 24 hours after surgery and continued for 28 to 42 days following the procedure, depending upon the patient’s other risk factors. LMWH is advantageous in that it can be given at a constant dose without any laboratory monitoring. Randomized clinical trials comparing LMWH with unfractionated heparin in general surgical patients have found that LMWH given once or twice daily are as effective or more effective in preventing thrombosis [33,34]. Warfarin has also been compared with LMWH, and most studies show a superior benefit with LMWH [35,36]. The patient should be encouraged to start moving the lower extremities as soon as possible postoperatively. Intermittent sequential compression devices (SCDs) for prophylaxis against deep vein thrombosis (DVT) on the contralateral limb is important while the patient is still in the hospital.

Hardware failure in the form of wire breakage will occasionally occur, more frequently with the wires in the foot. This is usually because of excessive strain in the foot during walking with a fixed ankle [12]. If this occurs, the wire can be removed and replaced with a new wire if necessary. It is important to check and adjust tension of all wires at postoperative visits. For patients over 200 pounds, it is recommended to place three wires across the tibia at each level of the tibial rings. This will aide in prevention of hardware failure.

It is important to check for ligamentous laxity and instability immediately after the frame is removed intra-operatively. If ligament damage is present, repair of the lateral ankle ligaments is often necessary. If the patient is complaining of symptoms related to this condition postoperatively when the frame is removed, these symptoms can usually be managed successfully with physical therapy and functional bracing.

Failure of the ankle distraction procedure to relieve pain is always a possibility. Ankle arthrodiatasis is generally performed on patients who have advanced, debilitating arthritis, and the patient must go into the procedure with the understanding that an arthrodesis or an implant may be necessary in the future. After ankle distraction there is often a period of increased pain and stiffness for two to four months, and it can take as long as six to twelve months to see improvement. During this time, the patient should continue with aggressive physical therapy and non-impact activities [31]. Radiographic improvements are sometimes seen for as long as five years after arthrodiatasis, indicating that ankle distraction benefits are progressive in nature [12]. It is the author’s belief that the benefit of ankle arthrodiastasis outweighs the mild and infrequent nature of the complications encountered with this procedure.

Ankle distraction with ankle arthroplasty should be considered a viable treatment for severe ankle arthritis for its minimal dissection and joint-sparing properties. Future treatment for ankle arthritis will likely involve ankle distraction in conjunction with newer methods in cartilage repair, such as autologous chondrocyte transplantation, autologous osteochondral transfer, and allografts. With new advances and developments, further studies will be required to study the efficacy of these procedures.

References

1.  Thomas RH, Daniels TR. Ankle arthritis. JBJS 2003 85A: 923-936.  [PubMed]
2.  Saltzman CL, Salamon ML, Blanchard GM, Hayes A, Buckwalter JA, Amendola A..  Epidemiology of ankle arthritis: Report of a consecutive series of 639 patients from a tertiary orthopaedic center. Iowa Orthop J 2005 26:44-46. [PubMed]
3.  Salk, RS, Chang TJ, D’Costa WF, Soomekh DJ, Grogan KA. Sodium hyaluronate in the treatment of osteoarthritis of the ankle: a controlled, randomized, double-blind pilot study. JBJS J 2006 88A:  295-302. [PubMed]
4.  Coester LM, Saltzman CL, Leupold J, Pontarelli W. Long-term results following ankle arthrodesis for post-traumatic arthritis.  JBJS 2001 83A: 219-228. [PubMed]
5.  Muir DC, Amendola A, Saltzman CL. Long-term outcome of ankle arthrodesis. Foot Ankle Clin. 2002 Dec;7(4):703-708. [PubMed]
6.  Thomas R, Daniels TR, Parker K. Gait analysis and functional outcomes following ankle arthrodesis for isolated ankle arthritis.  JBJS 2006 88A: 526-535.[PubMed]
7.  Knecht SI, Estin M, Callaghan JJ, Zimmerman MB, Alliman KJ, Alvine FG, Saltzman CL. The agility total ankle arthroplasty. Seven to sixteen-year follow-up. JBJS 2004 86A: 1161-1171. [PubMed]
8.  Spirit AA, Assal M, Hansen ST Jr. Complications and failure after total ankle arthroplasty. JBJS 2004 86A: 1172-1178.[PubMed]
9.  Paley D, Lamm BM. Ankle joint distraction. Foot Ankle Clin 2005 10: 685-698. [PubMed]
10.  Volkov MV, Oganesian OV. Restoration of function in the knee and elbow with a hinge-distractor apparatus. JBJS 1975 57A:591-600. [PubMed]
11.  Judet R, Judet T. The use of a hinge distraction apparatus after arthrolysis and arthroplasty. Rev Chir Orthop Reparatrice Appar Mot. 1978 64:353-365. [PubMed]
12.  Marijnissen AC, van Roermund PM, van Melkebeek J, Lafeber FP. Clinical benefit of joint distraction in the treatment of ankle osteoarthritis. Foot Ankle Clin 2003 8: 335-346.
13.  Marijnissen AC, van Roermund PM, van Melkebeek J, Schenk W, Verbout AJ, Bijlsma JW, Lafeber FP. Clinical benefit of joint distraction in the treatment of severe osteoarthritis of the ankle: proof of concept in an open prospective study and in a randomized controlled study. Arthritis Rheum 2002 46: 2893-902.
14.  van Roermund PM, Marijnissen AC, Lafeber FP.Joint distraction as an alternative for the treatment of osteoarthritis. Foot Ankle Clin 2002 7: 515-527. [PubMed]
15.  van Valburg AA, van Roermund PM, MarijnissenAC, van Melkebeek J, Lammens J, Verbout AJ, Lafeber FP, Bijlsma JW. Joint distraction in treatment of osteoarthritis: a two-year follow-up of the ankle. Osteoarthritis Cartilage 1999 7: 474-479. [PubMed]
16.  van Roermund PM, van Valburg AA, Duivemann E, van Melkebeek J, Lafeber FP, Bijlsma JW, Verbout AJ.Function of stiff joints may be restored by Ilizarov joint distraction. Clin Orthop Relat Res 1998 348: 220-227. [PubMed]
17.  van Valburg AA, van Roermund PM, Lammens J, van Melkebeek J, Verbout AJ, Lafeber EP, Bijlsma JW.  Can Ilizarov joint distraction delay the need for an arthrodesis of the ankle?  A preliminary report. JBJS 1995 77B: 720-725.[PubMed]
18.  van Valburg AA, van Roermund PM, Marijnissen AC, Wenting MJ, Verbout AJ, Lafeber FP, Bijlsma JW. Joint distraction in treatment of osteoarthritis (II): effects on cartilage in a canine model. Osteoarthritis Cartilage 2000 8:1-8. [PubMed]
19.   Krause FG, Windolf M, Bora B, Penner MJ, Wing KJ, Younger AS. Impact of complications in total ankle replacement and ankle arthrodesis analyzed with a validated outcome measurement. JBJS 2011 93A: 830-839. [PubMed]
20.  Kitaoka HB, Alexander IJ, Adelaar RS, Alexander IJ, Nunley JA, Myerson MS, Sanders M. Clinical rating systems for the ankle-hindfoot, midfoot, hallux and lesser toes. Foot Ankle Int. 1994 15: 349-353.[PubMed]
21.  Paley D. The principles of deformity correction by the Ilizarov technique: technical Aspects. Tech Orthop 1989 4:15-29.
22.  Mann R. Arthrodesis of the foot and ankle. In Surgery of the Foot and Ankle. Editors: Mann R, Coughlin M, St Louis, 1999,  Mosby, 651-698.
23.  Chiodo CP, McGarvey W.  Joint distraction for the treatment of ankle osteoarthritis. Foot Ankle Clin 2004 9: 541-553. [PubMed]
24.  Ploegmakers JJ, van Roermund PM, van Melkebeek J, Lammens J, Bijlsma JW, Lafeber FP, Marijnissen AC. Prolonged clinical benefit from joint distraction in the treatment of ankle osteoarthritis. Osteoarthritis Cartilage 2005 13: 582-588.[PubMed]
25.  Cole AA, Marqulis A, Kuettner KE. Distinguishing ankle and knee articular cartilage. Foot Ankle Clin 2003  8: 305-316. [PubMed]
26.  Schäfer DB. Cartilage repair of the talus. Foot Ankle Clin. 2003 8: 739-749. [PubMed]
27.  Treppo S, Koepp H, Quan EC, Cole AA, Kuettner KE, Grodzinsky AJ. Comparison of biomechanical and biochemical properties of cartilage from human knee and ankle pairs. J Orthop Res 2000 18:739-748. [PubMed]
28.  Egol KA, Paksima N, Puopolo S,  Klugman J, Hiebert R, Koval KJ.   Treatment of external fixation pins about the wrist: a prospective, randomized trial.  JBJS 2006 88A: 349-354. [PubMed]
29.  Davies R, Holt N, Nayagan S. The care of pin sites with external fixation. JBJS 2005 87B: 716-719. [PubMed]
30.  Paley D. The correction of complex foot deformities using Ilizarov’s distraction osteotomies. Clin Orthop Relat Res 1993 (293): 97-111. [PubMed]
31.  Beaman DN, Gellman RE, Trepman E. Deformity correction and distraction arthroplasty for ankle arthritis. Tech Foot Ankle Surg 2006 5:134-143.
32.  Rozbruch RS. Posttraumatic reconstruction of the ankle using the Ilizarov method. HSS J 2005 1: 68-88. [PubMed]
33.  Kakkar VV, Cohen AT, Edmonson RA, Phillips MJ, Cooper DJ, Das SK, Maher KT, Sanderson RM, Ward VP, Kakkar S.. Low molecular weight versus standard heparin for prevention of venous thromboembolism after major abdominal surgery. Lancet 1993 341: 259-265. [PubMed]
34.  Nurmohamed MT, Rosendaal FR, Buller HR, Dekker E, Hommes DW, Vandenbroucke JP, Briët E. Low-molecular-weight heparin versus standard heparin in general and orthopaedic surgery: a meta-analysis. Lancet 1992 340:152-156. [PubMed]
35.  Francis CW, Pellegrini VD Jr, Totterman S, Boyd AD Jr, Marder VJ, Liebert KM, Stulberg BN, Ayers DC, Rosenberg A, Kessler C, Johanson NA. Prevention of deep-vein thrombosis after total hip arthroplasty. Comparison of warfarin and dalteparin. JBJS 1997 79A:1365-1372. [PubMed]
36.  Hull RD, Pineo GF, Francis C, et al.  Low-molecular-weight heparin prophylaxis using dalteparin in close proximity to surgery vs warfarin in hip arthroplasty patients: a double-blind, randomized comparison. The North American Fragmin Trial Investigators. Arch Intern Med 2000 160: 2199-21207. [Website]


Address correspondence to: Craig Clifford, DPM, Federal Way Orthopedic Associates, Federal Way, WA 98003
Email: CraigClifford@fhshealth.org

1Director: Chicago Foot & Ankle Deformity Correction Center; 233 East Erie, Ste 702, Chicago, Illinois 60611
2Director: Franciscan Foot & Ankle Institute; 34509 9th Ave S., Ste 306, Federal Way, WA 98003.
3Federal Way Orthopedic Associates, Federal Way, WA 98003.
4St. Cloud Orthopedics, St. Cloud, Minnesota.

© The Foot and Ankle Online Journal, 2012

Correction of Traumatic Ankle Valgus and Procurvatum using the Taylor Spatial Frame: A Case Report

by Thurmond Lanier DPM, MPH , Erik Lilja DPM, FACFAS 

The Foot and Ankle Online Journal 4 (5): 3

Injuries about the ankle joint can have devastating consequences when left untreated or undertreated, and treatment is especially important in the pediatric population. Physeal injury may occur that can result in abnormal growth patterns. External fixation can be used to correct ankle and tibial deformities, and the Taylor Spatial Frame (TSF) can be used to more easily correct triplane deformities. A case study is presented to demonstrate the use of the TSF in correction of ankle valgus and tibial procurvatum.

Key words: Epiphysis, Ilizarov frame, External fixation, CORA

Accepted: April, 2011
Published: May, 2011

ISSN 1941-6806
doi: 10.3827/faoj.2011.0405.0003


Injuries about the ankle joint such as fractures, chronic ligamentous injuries, and osteochondral lesions can result in post-traumatic arthritis. These injuries are more devastating when they are left untreated or undertreated. When injuries of this nature happen in the pediatric population, special considerations must be taken to preserve physeal growth plates and prevent the occurrence of future growth disturbances. When a disturbance does occur at the epiphysis, depending how severe the deformity, surgical correction may be indicated. Internal fixation may be used to accomplish correction, but external fixation may be preferred if the deformity is severe which may result in soft tissue compromise if corrected acutely.

Ilizarov circular frames have had widespread use in the correction of angular deformities. [1] With more complex deformities (such as those in multiple planes), Ilizarov frames may be used but the application of hinges are needed and more complex pre-operative planning as well as more intricate post-operative manipulation is needed. With the advent of the Taylor Spatial Frame (TSF), deformities can be reduced in all three planes simultaneously. The TSF has two rings and six struts that are expandable. [2] The deformity parameters are measured by the surgeon and are introduced into the TSF software in which a prescription is printed. [3] We present a case report of a pediatric case where a TSF was utilized for correction of a multiplanar ankle deformity.

Case Report

A 14 year-old patient presented to clinic with complaints of right ankle pain. The patient related having a history of a right ankle fracture which happened 4 years prior. The patient states that he was making a jump on his quad and ended up catching the right foot on the top of the rear tire and felt a snap. The patient was diagnosed with an ankle fracture and was treated conservatively in a cast for approximately six weeks. The patient removed his own cast at that time and began weight bearing. The patient continued to have lateral ankle pain and noticed that his foot was in an abnormal position relative to his ankle. Radiographs were obtained 2 years after the initial injury which showed collapse of the lateral portion of the epiphysis and shortening with lateral displacement of the fibula with an increase in medial clear space. (Figs. 1A and 1B) This also resulted in valgus position of the ankle joint. The patient was given conservative treatment for approximately two years until his growth plates were closed and operative intervention was then initiated.

 

Figures 1A and 1B  Lateral pre-operative radiograph displaying tibial procurvatum and relatively narrow joint space. (A)  The anterior posterior (AP) and mortise pre-operative radiographs demonstrating significant valgus of the mortise and increased medial clear space. Also note decreased tibia-fibula overlap. (B)

Operative Technique/Post-operative care

The TSF consisted of two tibial rings connected by six struts. The proximal ring was applied to the tibia via two half pins. The half pins were applied perpendicular to the posterior crest of the tibia and confirmed with fluoroscopy. The distal ring was placed with two wires and a half pin and was placed distal to where the proposed osteotomy would be. The distal ring was placed in relative malalignment with the distal tibia so when the deformity was corrected the frame would be in a neutral position. An incision was then made along the anteromedial aspect of the tibia. Layered dissection was taken down to the tibia shaft and a through and through osteotomy was made with a sagittal saw. The osteotomy was made as distal in the tibia shaft as possible as the center of rotation of angulation (CORA) was located within the ankle joint. The CORA represents the apex of the deformity and in most cases is the optimal location to place the osteotomy.

If the CORA is located within a joint, generally the osteotomy is made proximal to the CORA within bone with good blood supply. A lateral incision was then made over the fibula with dissection taken down to the shaft. Utilizing a sagittal saw a transverse osteotomy was made. The final frame construction was made sure to be orthogonal to the tibia. (Fig. 2) Post-operatives radiographs were taken to ensure proper alignment of the frame. (Figs. 3 and 4) Adjustments were started approximately 10 days after surgery. Radiographs were taken on a weekly basis. The only complication that occurred was a pin tract infection which resolved with antibiotics. The frame was removed in 3 months when bony consolidation of the osteotomy was identified on radiographs. (Figs. 5 and 6)

Figure 2  Intra-operative image showing application of the Taylor Spatial Frame (TSF).

Figure 3  Immediate AP post-operative radiograph. Note distal ring with built-in deformity to match deformity at ankle joint.

Figure 4   Immediate lateral post-operative radiograph showing fibular osteotomy.

Figure 5  Final lateral post-operative radiograph showing healed osteotomy sites and decrease tibial procurvatum.

Figure 6  Final AP post-operative radiograph showing healed osteotomy sites with decrease in valgus position of the ankle.

Discussion

The use of the TSF has been described in numerous cases in the literature. The frame has been utilized in accomplishing ankle arthrodesis in patients with and without ankle and tibial deformities. Thiryayi, et al., described ankle fusion using the TSF in 10 patients. [2] The patients stayed in the fixator for an average of 24 weeks.

All patients demonstrated bony union. They reported having 7 cases of superficial pin tract infections which were resolved with oral antibiotics. Tellisi, et al., describe applying a TSF for limb lengthening and ankle fusion simultaneously. [4] The authors applied the TSF for ankle fusion and then brought patients back to the operating room for tibial lengthening. The surgeons performed the tibial osteotomy just distal to the tibial tuberosity. The authors also performed a fibular osteotomy to prevent tethering and angular deformity at the tibial lengthening site. The authors had 53 ankle fusions in which 12 underwent simultaneous tibial lengthening. 84% went on to complete fusion with two patients having significant non-unions (these patients were smokers). The average time in the fixator was 8.4 months with all patients having completely healed osteotomy sites. No significant pin tract infections were reported.

A case study was reported by Mabit, et al., where a TSF was placed on a young girl with ankle varus that resulted from a malunited ankle fracture. [5] The authors chose to correct her deformity gradually with a TSF preassembled and with the tibial rings oriented 30 degrees in the coronal plane matching the deformity. A tibial osteotomy was performed and distraction took place on the fourth post-operative day. The patient stayed in the TSF for 2.5 months. The patient went on to successful healing.

Feldman, et al., describe using the TSF for tibial malunions and nonunions. They had 18 patients in their study that had a TSF applied. The average time in the frame was 18.5 weeks. All patients went on to successful healing except one who developed a varus deformity through the healing fracture in the tibia. Fifteen of the 18 patients returned to preinjury activities at last follow-up. [3] Matsubara, et al., describe application of a TSF for 3 patients due to ankle ankylosis. [6] All patients had limb length discrepancy and angulation deformity. The average time in the fixator was 216 days. All patients were able to walk normally with a plantigrade foot.

Conclusion

The TSF is a useful tool for the surgeon to correct complex multiplane deformities of the lower extremity. Pre-operative parameters combined with the computer software make the TSF a simpler system as compared to traditional external fixators. There are various studies in the literature that demonstrate the usefulness and ease of this technique. Our patient was able to adjust the frame with ease and deformity correction was more precise using the computer software.

References

1. Chaudhary M. Taylor spatial frame-software-controlled fixator for deformity correction – The early Indian experience. Indian J Orthop 2007 41: 169-174.
2. Thiryayi WA, Naqui Z, Khan SA. Use of the Taylor Spatial Frame in Compression arthrodesis of the ankle: A study of 10 cases. J Foot Ankle Surg 2010 49: 182-187.
3. Feldman DS, Shin SS, Madan S, Koval KJ. Correction of tibial malunion and nonunion with six-axis analysis deformity correction using the Taylor Spatial Frame. J Orthop Trauma 2003 17: 549-554.
4. Tellisi N, Fragomen TA, Ilizarov S, Rozbruch SR. Limb salvage reconstruction of the ankle with fusion and simultaneous tibial lengthening using Ilizarov/Taylor Spatial Frame. Hospital Special Surgery, 2008 4:32-42.
5. Mabit C, Pecout C, Arnaud JP. Ilizarov’s technique in correction of ankle malunion. J Orthop Trauma 1994 8: 520-523.
6. Matsubara H, Tsuchiya H, Takato K, Tomita K. Correction of ankle ankylosis with deformity using the Taylor Spatial Frame: A report of three cases. Foot Ankle Int 2007 28: 1290-1294.


Address correspondence to: Thurmond Lanier, DPM, MPH, Swedish Medical Center, 747 Broadway, Seattle, WA, 98122.

1  PGY 2, Swedish Medical Center, 747 Broadway, Seattle, WA, 98122
2  Attending physician, Swedish medical center, 747 Broadway, Seattle, WA, 98122, Private practice, 9501 5th Ave. NE, Seattle, WA, 98115.

© The Foot and Ankle Online Journal, 2011

Talar and Calcaneal Y-Osteotomy with Distraction Osteogenesis for the Correction of Rigid Equinus

by Sutpal Singh, DPM, FACFAS , Albert Kim, DPM 2, Timothy Dailey, DPM 3, Long Truong, DPM 4, Maria Mejia, DPM 5

The Foot and Ankle Online Journal 4 (4): 3

Types of equinus, surgical management for equinus involving the Y- talocalcaneal osteotomy supplementing an external fixation device is presented. A case report is introduced involving surgical correction of a 53 year old male who had a severe equinus flat top talus with mild varus secondary to clubfoot surgery. Treatment included surgical correction utilizing Steindler stripping, Achilles tendon lengthening, and a rather rare Y- osteotomy of the calcaneus and talus with the use of a multiplaner external fixator in an unconstrained system to correct the equinus and varus deformity. Slow distraction was performed in order to decrease the risks of having neurovascular injury, soft tissue injury, and shortening of the foot. After months of follow-up, there was good healing of the osteotomy sites and the patient had a plantigrade foot.

Key words: Clubfoot, Rigid Equinus, Flat top talus, Y-Osteotomy, External fixation, Distraction Osteogenesis, Ilizarov method.

Accepted: March, 2011
Published: April, 2011

ISSN 1941-6806
doi: 10.3827/faoj.2011.0404.0003


Ankle joint equinus can be divided into a three types; soft tissue, osseous and a combination of the two. [1] Soft tissue equinus alone can be easily repaired, but when multiple deformities occur such as clubfoot, it can complicate the treatment.

The etiology of ankle equinus can include trauma, diabetes mellitus, poliomyelitis, osteomyelitis, contracture from burns, neglected or relapsed clubfeet, prolonged immobilization in plantarflexion, as well as neuromuscular disease. [1,3,7]

Traditionally, correction has been managed through arthrodesis and extensive soft tissue release. Treatments for soft tissue ankle equinus include procedures such as splinting, tendo-Achilles lengthening, gastrocnemius recession, and rare deformities such as accessory soleus release. [10,13]  Neurovascular, soft tissue problems and shortening of the foot may occur with traditional techniques. Also, prior surgeries may make the surgical management of the foot deformities more difficult later in life.

An Ilizarov external fixator can be used to correct equinus and it can be applied in two ways; constrained and unconstrained. The constrained technique places the axis of rotation at a planned anatomical axis of the joint. [9]  The constrained technique can correct deformities within the joint unlike the unconstrained that keeps the natural axis of rotation. The unconstrained technique places the axis of rotation around the natural axes of the joint. [9] The unconstrained technique requires distraction of the ankle joint before an attempted correction.

The Ilizarov external fixator can be used to correct equinus without an osteotomy, only when the equinus is soft tissue in nature. Only correcting the soft tissue deformity with an Ilizarov frame has a high recurrence rate. [3]  When dealing with an osseous equinus an osteotomy must be performed and Ilizarov used the process called distraction osteogenesis to correct the deformity. [18] “Gradual distraction of the soft tissues and bones enables reshaping of the foot.” [8]  The Ilizarov technique can fix these deformities with minimal incisions. The downside to this is the patient has an external fixator for several months. During this time the patient may bear weight on the area which has an advantage over internal fixation. Another advantage is during osseous correction it can also correct for soft tissue deformities at the same time.

There are three main types of Ilizarov rearfoot osteotomies including the U, V and Y osteotomies. [9]  The Y- osteotomy is the least published of the three. The Y is an osteotomy through the neck of the talus and an osteotomy of the posterior calcaneus below and parallel to the subtalar joint making an apex in the anterior calcaneus and then an osteotomy plantarly thus creating the shape of a “Y”. [9]  The Y- osteotomy has the same implications as the V- osteotomy except it doesn’t elongate the foot as much. [9] The Y- osteotomy with external fixation allows small changes each day to allow the new bone to form and correct the deformity. This osteotomy allows correction of complicated equinus cases. Which osteotomy is chosen depends greatly on the surgeons’ experience.

Case Report

A 53 year old male was seen in the office due to severe pain around the sub right 4th and 5th metatarsals. The patient had an equinus deformity of the right lower extremity with varus deformity. (Figs.1A and 1B) His past surgical history was significant for club foot surgery when he was 4 years old, which still left him ambulating on the ball of his right foot. The rest of the history was unremarkable.

 

Figure 1A and 1B  Preoperative photo (A) and  radiograph (B) showing ankle equinus deformity.

Physical exam showed that he had scar tissue around the medial, lateral, and posterior heel on the right foot. The positioning of his right lower extremity included a tight contracted Achilles tendon that correlated with a rigid equinus deformity, varus of the foot with subtalar joint stiffness with no range of motion at the mid foot, rear foot and ankle. Also, there were contractures of all the digits. There was no leg length discrepancy noted and the length of the foot was within normal limits. The patient had difficulty wearing shoes and could only wear sandals. There was increased pain under the right 4th and 5th metatarsophalangeal joints due to the excessive pressure while ambulating secondary to the rigid equinus and inversion of the foot. This, in effect, was placing too much pressure on his 4th and 5th metatarsal heads.

The procedure that was performed included a Steindler stripping, an Achilles tendon lengthening, talar and calcaneal Y-osteotomies, plantar tenotomy of all five digits, and the application of an Ilizarov external fixator.

The patient was prepped and draped in the usual standard manner. No tourniquet was applied. A # 15 blade was used to perform a percutaneous tendon lengthening of the Achilles Tendon. The foot was only able to dorsiflex several degrees due to the severity of the deformity from negative 45 to negative 40 degrees. Along the plantar medial calcaneus a Steindler stripping was then performed. This helped release all the fascial and muscular contractions on the plantar foot from the calcaneus. A prophylactic tarsal tunnel release was also performed by transecting the laciniate ligament. Two tibial rings were then applied to the lower leg. One half-ring was applied to the calcaneus and one half-ring was applied to the distal foot. All wires were then tensioned appropriately. The calcaneal half-ring was attached to the posterior tibial ring. The distal foot half-ring was then connected to the anterior tibial ring.

The calcaneal ring was then connected to the distal foot half ring. All connections had distraction capability to correct the foot in three dimensions. Prior to any distraction, using flouroscan a small incision was made at the medial aspect of the talar neck. The incision was deepened to the subcutaneous tissue and then to the periosteum of the talus. A small osteotome was then used to perform a complete osteotomy of the talar neck. Along the lateral aspect of the calcaneus a small incision was made parallel and inferior to the subtalar joint. The incision was deepened to the subcutaneous level and then to the periosteum of the calcaneus. A small osteotome was used to perform a complete osteotomy of the calcaneus 1 cm posterior and inferior to the subtalar joint. Along the anterior aspect of the calcaneus a small incision was made 1 cm proximal to the calcaneal cuboid joint. The incision was deepened to the subcutaneous level and then down to the periosteum of the calcaneus. A small osteotome was inserted down to bone and rotated being careful not to transect the peroneal tendons or sural nerve. A complete osteotomy was then performed at the distal calcaneus. (Fig. 2)

Figure 2  Intra-operative  radiograph of the  Y- osteotomy.   The left side of the Y is the Talar neck osteotomy, the right side of the Y is the calcaneal osteotomy below the subtalar joint and the stem of the Y osteotomy is located at the distal calcaneus.

Note that a void was created while removing the osteotome at the stem of the osteotomy. This eventually healed well due to the vascular nature of the calcaneus. All surgical sites were irrigated with normal saline and bacitracin and sutured with 3-0 Prolene. Attention was then directed to all the plantar toes and plantar flexor tenotomies were then performed and smooth wires were then inserted and attached to the distal half ring. The surgical site was then dressed with Adaptic, gauze and Kerlex . (Figures 3,4,5A, 5B, and 6)

Figure 3  Initial frame placement at the time of the surgery.

Figure 4  Note the rod rotating the distal ring out of varus and dorsiflexing it.

 

Figure 5A and 5B  Note that the forefoot and rearfoot can be manipulated independent of each other.  Note the wires from the toes being attached to the forefoot half ring. (A)  The plantar view showing the percutaneous plantar tenotomies of all the toes with wires going into the metatarsals and attached to the foot half ring. (B)

Figure 6  The post-operative radiograph.

At one week, the following manipulations to the bones in the external fixator were initiated: 1) distraction of the calcaneus towards eversion and inferior displacement, 2) rotation of the distal foot towards eversion and dorsiflexion, 3) distraction of the forefoot from the rearfoot.

The patient was instructed to increase the movements by a total of 1 mm per day achieved by a ¼ turn of the distraction mechanism 4 times per day. The patient was taken back to the OR at week 3 (Fig. 7) and at week 8 (Fig. 8) for the addition of extra rods and bars for greater manipulation and a better line of pull. At week 12, there was good alignment of the foot in a plantigrade position and at week 20, the external fixator was removed (Fig. 9) At 6 months, the patient was ambulating in a custom made AFO. (Fig. 10)

Figure 7   More rods were applied for more distraction at 3 weeks after surgery.

Figure  8   At 8 weeks , the patient had more bars and rods added with frame manipulation.

Figure 9  At 5 months the frame was removed and the patient was placed in a below the knee cast.

Figure 10   At 6 months, the patient had his foot plantigrade and was full weight bearing in a custom made AFO.

Overall, the patient was able to ambulate with the heel on the ground with minimal pain; however, did not have much range of motion at the ankle or foot. All the pain under the lateral forefoot resolved. The patient was satisfied with his plantigrade foot and able to ambulate with a custom AFO.

Discussion

There are multiple surgical procedures available for correction of acquired ankle equinus, soft tissue as well as bone procedures. [1,2,4] When this deformity becomes fixed it poses a challenge to many foot and ankle surgeons due to soft tissue contraction and bony adaption and requires a combination of soft tissue and bone procedures. [1,2,4] Flexible deformities can be treated with manipulation methods thus preventing surgery.

When manipulation methods don’t work then surgery is required. Surgical correction consisting of extensive soft tissue releases with and without arthrodesis for equinus deformity has been well described. [15] Different osteotomies with and without an external fixator have been described in literature for correction of complex foot deformities. [9,11,14,16,17,20]

Correcting deformities such as clubfoot become a challenge especially after failed surgeries due to stiffness of the soft tissues and residual deformities such as equinus. [16,17] There are several osteotomies and a few of the commonly discussed ones are the U (Scythe-shaped), V, and less commonly discussed Y- osteotomy. [16,17]

The scythe-shaped (U) osteotomy is a curved osteotomy that divides the foot into two sections. It starts posterior to the lateral malleolus and runs from 1-1.5 cm below the posterior subtalar joint, then penetrates the floor of the sinus tarsi and emerges at the talar neck. [9,11] This type of osteotomy allows for correction of equinus with a rigid tibio-talar joint. [9]

The V – osteotomy is a combination of oblique cuts that are angled at 60-70 degrees. [9] The osteotomy is performed along the posterior calcaneus and the anterior calcaneal-talar. This type of procedure is indicated for treatment of complex deformity of the hindfoot and the midfoot. [9,11,19,20] The V- osteotomy offers versatility when combined with an external fixator because it has the ability to preserve foot length and perform simultaneous tibial corrections. [20]

The Y – osteotomy is similar to the V – osteotomy in a sense that it allows one to apply differentiated correction between the hindfoot and the forefoot. The osteotomy results in a three ray star that is all 120 degrees apart. The osteotomy is first performed at the oblique posterior aspect of the calcaneus, then a vertical osteotomy of the calcaneus, and finally the calcaneal-talar ostetomy. [9] This type of procedure allows for the same correction as the V – osteotomy but with fewer complications. [9]

The hinges are positioned on the medial and lateral threaded rods of the calcaneal half ring. The equinus is corrected by lowering the calcaneus and raising the forefoot in relationship to the talar body. [9] Correction is achieved through the movement of the fragments of the osteotomy with the majority of correction of the calcaneal and talar equinus. [9,14] The Y – osteotomy does not cause any skeletal lengthening as with the scythe-shaped osteotomy, therefore it offers three advantages. [9] The advantages include faster consolidation because of less bone regeneration, skin alteration is easily contained, and prevention of calcaneocuboid diastasis is unnecessary. [9]

The Ilizarov method with external fixation was chosen for the correction after performing the osteotomy because it enables correction in all three orthogonal planes. [4,9,11,14,17] Using an external fixator is not only minimally invasive, but it also allows the surgeon to stage the treatment appropriately to manipulate the rate and direction of the correction. It can be used as either a constrained or unconstrained hinge system. In the constrained foot frame, forces applied to the foot are directed around the axis. [14] This technique is usually reserved for large joints. In the unconstrained system, joints of the ankle and the foot are used as the fulcrum points for correction and it is usually used with smaller joints or deformities with multiple joint axes. [14]

The Steindler stripping procedure is recommended for patient with significant contractures of the plantar aponeurosis and plantar musculature.[1] The abductor hallucis, flexor digitorum brevis, and abductor digiti quinti are released from the periosteum of the calcaneus. However, this procedure is limited in that it does not correct fixed deformities and only corrects in the sagittal plane. [1]

The complications associated with the use of an external fixator and any type of osteotomy includes tarsal tunnel syndrome, neurovascular symptoms, pin tract infection, flexor contractures, valgus drift, incomplete osteotomy, residual deformity, and recurrence of problem. [4,9,11,14,16,17,19,20]

Due to such complications, it has been recommended that a prophylactic tarsal tunnel release be performed to decrease the likelihood of nerve entrapment secondary to the correction and to minimize the risk of vascular injury. [16] There is a high incidence of nerve injury as a result of acute angular deformity correction. [20] If compressive symptoms of the tibial nerve are experienced during the correction, it can be addressed either by performing a secondary surgical decompression or by decreasing the rate of correction. [11,14] Pin tract infection and flexor contractures are usually secondary to prolonged fixator utilization. [19,20] Pin tract problem are related to skin motion and controlled with local pin site care. Pin tract infections are minor complications that occurs with any type of external fixator but respond very well to oral antibiotics and rarely lead to osteomyelitis requiring pin removal. [17,20] Premature consolidation of the osteotomy before full correction is reached is also not an uncommon complication. To avoid this problem, it is recommended to start distraction routinely on the third postoperative day. [16]

Despite such complications, the Ilizarov technique remains an effective and safe tool for complex lower limb reconstruction surgery. It allows corrections in all planes at a rate that can be tailored to the deformity without the constraints of traditional methods.

When looking back at the literature regarding the Y- osteotomy there is not much to be found. Our case presentation is unique that to our knowledge there are no other journal articles on which a y calcaneal osteotomy is used in conjunction with an Ilizarov distractor. Furthermore there is close to no literature regarding the Y- osteotomy. In the literature the main focus has been on correcting the structural equinus foot by using the V. [16,17] Also there has not been another study in which the y osteotomy is used in conjunction with the Ilizarov distractor. In our case study we found that the Y- osteotomy allows for correction of a severe deformity while minimizing neurovascular and soft tissue complications as well as avoiding excessive shortening of the foot as is many times encountered with traditional techniques.

Traditional methods although successful with certain patients they involve much more cut in the bone which can lead to excessive shortening and soft tissue complications. Our case report helps to illustrate a successful way in which a rigid equinus can be corrected by the use of an under researched osteotomy with gradual distraction of the structures in the foot. In our case the patient was satisfied with his plantigrade foot even though he did not have much range of motion at the ankle or foot. This view is supported by other studies in which patient satisfaction is achieved with improvement in appearance of the foot. [6,16,21]

Conclusion

In this case study, we presented a rigid equinus foot that was corrected with the use of a Y – osteotomy along with the use of Ilizarov methodology. There is not much literature about the usage of the Y – osteotomy even though it has three main advantages which are: faster consolidation, skin alterations are easier to contain, and there is less chance of calcaneal-cuboid diastasis. [1] Furthermore, the Y – osteotomy avoids excessive lengthening of the foot. Correction of severe foot deformities with the Ilizarov method is technically difficult but when used with the Y – osteotomy, differentiated correction between the hindfoot and forefoot can be applied. In the case study it was successfully shown that the Y – osteotomy allows for correction of a severe deformity while minimizing neurovascular and soft tissue complications as well as avoiding excessive shortening of the foot as is many times encountered with traditional techniques. The final result was a plantigrade foot. Thus, the Y – osteotomy through the talus and calcaneus with distraction osteogenesis using the Ilizarov methodolgy is an effective surgical procedure in correcting rigid equino-varus foot deformities.

References

1. Banks AS, Downey MS, Martin DE, Miller SE. McGlamry’s Comprehensive Textbook of Foot and Ankle Surgery. Philadelphia, Lippincott Williams & Wilkins, 2001.
2. Coughlin, Michael J, Roger A. Mann, Charles L. Saltzman. Surgery of the Foot and Ankle. Philadelphia, Mosby 2007.
3. Digiovanni CW, Holt S, Czerniecki JM, Ledoux WR, Sangeorzan BJ. Recurrence after correction of acquired ankle equinus deformity in children using Ilizarov Technique. Strategies Trauma Limb Reconstruction 3: 105-108, 2008.
4. Emara Khaled M, Allam Mohamed Farouk, ElSayed Mohamed Nabil MA, Ghafar Khaled Abd EL. Recurrence after correction of acquired ankle equinus deformity in children using Ilizarov technique. Strat Traum Limb Recon 3:105-108, 2008.
5. Easley, Mark E., Wiesel Sam. Operative Techniques In Foot and Ankle Surgery. Philadelphia, Lippincott Williams & Wilkins 2011.
6. Freedman JA, Watts H, Otsuka NY. The Ilizarov method for the treatment of resistant clubfoot: is it an effective solution? J Pediatr Orthop 26:432–437, 2006.
7. Guyton G, Saltzman C. The Diabetic foot. JBJS 83A: 1083-1096, 2001.
8. Ilizarov GA. Transosseous osteosynthesis. Berlin/Heidelberg: Springer-Verlag, 1992.
9. Kirienko Alexander, Villa Angelo, Calhoun Jason H. Ilizarov Technique for Complex Foot and Ankle Deformities. Marcel Dekker, Inc, 2004.
10. Kishta WE, Mansour EH, Ibrahim MM. The accessory soleus muscle as a cause of persistent equinus in clubfeet treated by the Ponseti method : A report of 16 cases. Acta Orthopaedica Belgica 76: 658-662, 2010.
11. Kocaoğlu M, Eralp L, Atalar AC, Bilen FE. Correction of complex foot deformities using the Ilizarov external fixator. J Foot Ankle Surg 41: 30-39, 2002.
12. Laughlin RT, Calhoun MD. Ring fixators for reconstruction of traumatic disorders of the foot and ankle. Orthop Clin North Am 287-294, 1995.
13. Lopez A, Kalish S, Mathew J, Willis FB. Reduction of ankle equinus contracture secondary to diabetes mellitus with dynamic splinting. Foot Ankle Online Journal. 3 (3):2, 2010.
14. Mendicino RW, Murphy L J, Maskill MP, Catanzariti AR, Harry P. Application of a constrained external fixator frame for treatment of a fixed equinus contracture. J Foot Ankle Surg 47: 468-475 , 2008.
15. Galli M , Cimolin V, Crivellini M, Albertini G. Gait analysis before and after gastrocnemius fascia lengthening in children with cerebral palsy. J Appl Biomaterials Biomechanics 3: 98-105, 2005.
16. Segev E, Ezra E, Yaniv M, Wientroub S, Hemo Y. V Osteotomy and Ilizarov technique for residual idiopathic or neurogenic clubfeet. J Orthopaedic Surg 16: 215-219, 2008.
17. Shalaby H, Hefny H. Correction of complex foot deformities using the V-osteotomy and the Ilizarov technique. Strat Traum Limb Recon 1: 21-30, 2007.
18. Spielberg Parratt Dheerendra Khan Jennings Marsh. Ilizarov principles of deformity correction. Annals of The Royal College of Surgeons of England 92: 101–105, 2010.
19. Gerhardt S, Vinay S, Bernhard ZE, Uitz C, Wolfgang L. Complex foot deformities associated with soft-tissue scarring in children. Journal Foot Ankle Surg 40: 42-49, 2001.
20. Theis JC, Simpson H, Kenwright J. Correction of complex lower limb deformities by the Ilizarov technique: An audit of complications. J Orthopaedic Surgery 8: 1448-1552, 2000.
21. Utukuri MM, Ramachandran M, Hartley J, Hill RA. Patient-based outcomes after Ilizarov surgery in resistant clubfeet. J Pediatr Orthop B 15:278–84, 2006.


Address correspondence to: Sutpal Singh, DPM, FACFAS, Chief Ilizarov Surgical Instructor at Doctors Hospital West Covina, Fellow of the American College of Foot and Ankle Surgeons, Private practice in Southern California.

1 Chief Ilizarov Surgical Instructor at Doctors Hospital West Covina, CA.  Fellow American College of Foot and Ankle Surgeons. Private practice in Southern California
2 PM&S 36, R3 (Foot and Ankle Medicine and Surgery) Doctors Hospital of West Covina.
3 PM&S, R2 (Foot and Ankle Medicine and Surgery) Doctors Hospital of West Covina.
4 PM&S, R1 (Foot and Ankle Medicine and Surgery) Doctors Hospital of West Covina.
5 PM&S, R1 (Foot and Ankle Medicine and Surgery) Doctors Hospital of West Covina.

© The Foot and Ankle Online Journal, 2011

Reconstructing Limb Deformities using the VCAM™ External Fixator: A series of 3 cases

by Michael P. DellaCorte, DPM, FACFAS , Panagiotis Panagakos, DPM ,
Tarika Singh, DPM , Howard Goldsmith, DPM, AACFAS

The Foot and Ankle Online Journal 4 (1): 2

External fixation was used almost exclusively for fracture management. It is also used for arthrodesis, management of lower extremity deformities such as Charcot neuroarthropathy, limb lengthening, osteotomy stabilization, osteomyelitis, nonunion or pseudoarthrosis. It has proven extremely useful in the treatment of a number of conditions because it can provide distraction, compression, stabilization and neutralization as needed. Traditional external fixators involve driving pins through the tibia and fibula. The VCAM™ is a unique below the ankle external fixator. The VCAM™ can avoid possible disruptions and complications that are often seen with traditional Ilizarov fixators. The indications for the VCAM™ external fixator are identical to the Ilizarov fixators, such as off-loading, fracture reduction and reconstructive procedures. In our institution, we have used the VCAM™ device to off-load ulcerations and correct limb deformities. In the cases presented in this paper the VCAM™ was used to off-load wounds secondary to Charcot arthropathy and transmetatarsal amputations, as well as to gradually correct a rearfoot deformity such as seen in a Chopart’s amputation. The VCAM™ can be constructed into an Ilizarov type frame or a hybrid frame which can be used to achieve gradual triplanar correction. We have seen good results using the VCAM™ for wound care and limb deformities and recommend this approach when tibia and fibula intervention is not necessary.

Key words: Limb deformity, Charcot foot, ulceration, VCAM™, wound, external fixation.

Accepted: December, 2010
Published: January, 2011

ISSN 1941-6806
doi: 10.3827/faoj.2011.0401.0002


External fixators have proven very versatile in treatment as they can be used with open, closed or limited open surgical techniques. They provide access to the involved limb for wound healing and dressing changes and can be designed to correct complex deformities such as Charcot joint. They allow for gradual, precise correction over the postoperative course rather than a single intra-operative correction by osteotomy or fusion.

Ring fixators are typically used to treat complex Charcot neuroarthropathy. [1] External fixators provide multiplanar correction including angulation, translation, rotation, stabilization, compression, distraction and neutralization while allowing for surrounding soft tissue adaptation; this also helps to minimize wound complications and vascular compromise that may result from overcorrection in a single-stage procedure. [2,4,5,6] In general, external fixators can be used to correct coexisting deformities separately, successively or simultaneously. [3]

The VCAM™ fixator allows for adjustment and alteration as needed during the post-operative period. Full immediate weight bearing postoperatively is also possible with the external fixator. [2] This is extremely important in Charcot reconstruction with concomitant ulcerations that require offloading. Additionally, external fixation is the only treatment option for Charcot with associated osteomyelitis or in Eichenholtz stage I and II, where internal fixation is contraindicated. [4]

There are two main types of fixators. The monolateral fixator which consists of threaded half-pins attached to a bar which allows for axial compression or distraction. The other main type is the ring fixator which was made popular by G.A. Ilizarov. The ring fixators use trans-osseous wires and pins placed under tension for bone fixation; they are more versatile and complex than the monolateral fixators. The hybrid fixator is a combination of these two main types and may be more appropriate for certain indications. [2,3]

Lower limb deformities secondary to trauma, diabetes or any other pathological cause can be devastating to patients and frustrate foot and ankle surgeons treating them. Charcot arthropathy is one of the limb deformities discussed in this paper that can lead to ulceration. Treatment of Charcot foot may require internal as well as external fixation. Ilizarov type fixators have been used for surgical reconstruction of this deformity. Surgery is indicated for treatment of Charcot arthropathy if chronic or recurrent ulcers are associated with the deformity, if the deformity is unstable and if there is an acute fracture in a neuropathic patient with good circulation. [1,2]

There is limited mention in the literature of external fixation devices that do not extend proximal to the ankle. The purpose of this paper is to introduce the VCAM™ external fixator and present its various uses and construct designs.

The VCAM™ has been available for nearly a decade. In our opinion it is an underutilized external fixator because it has not been previously reported in the literature which has lead to an ignorance of the device in the orthopaedic community.

It has mainly been used in locations where ankle privileges are not available to podiatrists. The VCAM™ consists of a leg portion with a boot construct similar to a CAM walker with Velcro straps lined along the leg sleeve, plastic upright Velcro extensions attached to posts, various size threaded rods, half rings, foot plates and a rocker bottom with rubber treads with Velcro straps. We believe this unique external fixator has many advantages over traditional Ilizarov frames. It reduces the number of pins or wires placed across the tibia and fibula, therefore decreasing pin site infections, calf edema does not become an issue, fractures of the tibia and fibula when inserting, removing and/or tensioning the wires and decrease in thermal necrosis of neurovascular and muscular structures in the leg. The VCAM™ cannot be used for limb lengthening procedures, ankle fractures, pilon fractures, or any other surgical procedure involving the tibia or fibula

Technique

When all ancillary pedal procedures are complete and the half rings and wires have been applied to the operative site and tensioned, the VCAM™ leg sleeve is first applied over cast-padding. Next, the plastic extensions are attached by velcro and are secured. Then three or four hole posts are secured to the plastic extensions to which various sized rods are attached spanning from the leg down to the foot. These rods are connected to the foot plate and half rings with the use of posts, if needed. Note that the foot plates and half rings are secured in place with nuts and bolts to the smooth olive transfixation wires during the surgical reconstruction. Lastly, the rubber rockerbottom foot attachment allows protection and partial weight bearing. Constructs are designed based on the pedal pathology present.

Once there is clinical and radiographic evidence of consolidation at the fusion site, or there is clinical correction of a specific limb deformity the external fixator may be removed. The frame is often removed after 8 to 12 weeks and the patient is fitted for a Charcot Restraint Orthotic Walker (CROW) and remains in this device for 6 months.

Thereafter, patients are fitted for ankle foot orthoses (AFO) and custom extra depth shoes with appropriate fillers if necessary.

Case Report 1

This diabetic male patient presented to the Emergency Department with an infected 2nd toe leading eventually to a proximal Chopart’s Amputation. The patient had multiple debridements and fasciotomies of the leg over a 6 month period. He developed a lateral ankle ulcer and an adducto-varus foot type as a result of muscle imbalances. (Figs. 1A and 1B)

Figure 1A and 1B Case 1: Lateral aspect of the lower extremity with adducto-varus deformity and ulceration over the fibular malleolus before VCAM™ application. (A)  A superior view of the lower extremity with adducto-varus deformity. (B)

He had the VCAM™ external fixator applied to offload the ulcer and to gradually correct the adducto-varus deformity. The pins were placed distally through the amputation site to create a more stable frame. He had an adjunctive Achilles tenotomy. Weekly adjustments consisted of tightening the lateral aspect of the frame and loosening the medial components to bring the foot perpendicular to the leg. (Figs. 2A, 2B and 2C) After six weeks of weekly adjustments clinical correction of the deformity was achieved and the lateral ulcer healed. The VCAM™ was then removed. (Figs. 3A and 3B) He was then placed in a CROW Walker to weight bear.

Figure 2A, 2B and 2C Case 1:  Day of VCAM™ application. (A)  1 week after the VCAM™ application with the first adjustment after VCAM™ application. (B)  4 weeks after VCAM™ application with the fourth adjustment. (C)

Figure 3A and 3B Case 1:  6 weeks after VCAM™ application and there is clinical correction of deformity.  The VCAM™ was then removed. (A) The lateral view shows clinical correction of deformity and healed ulceration. (B)

Even though the VCAM™ is primarily a below ankle frame it can be designed to imitate a Taylor Spatial™ external fixator, as it was for this case. A half ring was able to be applied above the ankle without any pins inserted into the leg and six struts were fashioned to help achieve gradual triplanar correction of the lower extremity deformity.

Case Report 2

A 60 year-old male with history of Diabetes Mellitus with peripheral neuropathy and ESRD, presented to wound care center with a chief complaint of chronic non-healing plantar ulcers of six months duration. The patient had a previous left foot trans-metatarsal amputation (TMA) with an ulcer on the distal plantar lateral aspect of the TMA site and a plantar heel ulcer. Local wound care with weekly debridements failed to heal the ulcers. The plantar heel ulcer measured 5cm x 6cm and probed to bone.

It was then decided to proceed with surgical debridement of the ulcer and VCAM™ application. On March 3rd, 2008 a percutaneous tendo-Achilles lengthening (TAL) and tenotomy of the anterior tibial tendon were performed to relieve forefoot pressure on the distal plantar lateral TMA site ulcer. The Versajet Hydrosurgery System™ (Smith & Nephew) was used to debride the plantar ulcers of all necrotic tissue and then application of Apligraf® (Organogenesis) skin substitute was applied to the heel ulcer. At this point, a VCAM™ external fixator was applied to offload the plantar ulcerations and help maintain angular correction after TAL and anterior tibialis tenotomy. The pins for the frame were thrown distally through the TMA site exiting posterior to the heel to help create a more stable construct. The VCAM™ in this case is a standard Ilizarov type frame and was primarily used to offload the ulcers. (Fig. 4A and 4B) The ulcers were progressing well and had decreased in size significantly until the patient tripped and fell while ambulating which ultimately led to several pin tract infections. (Figs. 5A and 5B) The causative organism of the pin site infections was MRSA. The patient was started on Zyvox® (Pfizer) and the VCAM™ fixator was removed on April 17, 2008.

Figure 4A and 4B Case 2:  Clinical appearance the day of VCAM™ application. (A) The lateral view 1 week after VCAM™ application. (B)

Figure 5A and 5B Case 2:  2weeks after VCAM™ application showing ulcer healing with associated pin tract infections. (A)  4 weeks after VCAM™ application with progressive closure of the ulcers.

The distal plantar ulcer healed before the fixator was removed and all wounds healed with continued off-loading after removal. Pin site infections are the most common complication with external fixators. In this case, patient selection was appropriate. He could ambulate without any significant issues prior to VCAM™ application that would deter a foot and ankle surgeon from applying an external fixator. In our opinion the result of the patient falling was accidental.

Case Report 3

A 51 year-old diabetic female with history of Hypertension, Hypercholesterolemia, Charcot Neuroarthropathy and a non-healing Wagner Grade 3 ulcer measuring 2.4 cm x 2.5 cm x 2.4 cm present for more than 1 year duration, was seen in the Wound Care Center. (Figs. 6A and 6B) Radiographs revealed a Charcot foot deformity with dislocation at the LisFranc and Chopart joints. (Figs. 7A and 7B) After 17 hyperbaric oxygen treatments helped to resolve cyanosis of the digits, it was decided that surgical intervention would be necessary to realign the midfoot and to offload the ulcer.

Figure 6A and 6B Case 3:  The Initial clinical appearance; plantar view of the foot. (A)  The Initial clinical appearance demonstrating the depth of the ulcer. (B)

Figure 7A and 7B Case 3:  The initial radiographic lateral view. (A) The initial radiographic dorsoplantar view. (B)

The patient had a wound debridement, left talar osteotomy, percutaneous Tendo-Achilles lengthening and VCAM™ application. This frame was constructed to offload the ulcer and to compress and realign the midfoot to the hindfoot. One half ring was placed on the dorsal aspect of the foot and another half ring placed posterior to the heel to aid with compression. Realignment of the dislocated joints is evident on the immediate post operative radiographs. The frame was adjusted on a weekly basis. The VCAM™ was successful in producing a more plantigrade foot and offloading the ulcer long enough for it to decrease greatly in size. The frame was removed after 8 weeks and the patient was subsequently put into an ankle foot orthosis. Conservative wound care continued for approximately 2 months until the ulcer healed successfully. (Figs. 8A, 8B and 8C)

Figure 8A, 8B and 8C Case 3: Day of VCAM™ application. (A)  The foot is placed in a more plantarflexory position to promote ulcer closure. (B)  The immediate post-op lateral radiograph. (C)

Discussion

External fixators are now almost exclusively used for arthrodesis, management of lower extremity deformities such as Charcot neuroarthropathy, limb lengthening, osteotomy stabilization, osteomyelitis, nonunion or pseudoarthrosis. [1,2] In wound and ulcer management it provides offloading and potentiates healing. It has proven extremely useful in treatment of these conditions because it can provide distraction, compression, stabilization and neutralization as needed. [2]

There is limited mention in the literature of external fixation devices that do not extend proximal to the ankle. Herbst uses two types of external fixation devices for the treatment of Charcot Arthropathy. One is a foot frame and the other a tibiocalcaneal frame. He uses the foot frame for the correction of midfoot deformity. The main characteristics are a hindfoot ring and a forefoot ring in the coronal plane. The two rings have a spanning device between them to provide compression across the midfoot. [7] Malizos, et al., described an Ilizarov below the ankle circular frame to treat displaced calcaneal fractures. There are 2 rings both confined to the foot.

The proximal ring serves a stable ground through the talus and midfoot bones and supports the distal ring. The 2 rings are distracted to withstand the deforming forces of the Achilles tendon, the plantar musculature, aponeurosis and peroneal retinaculum. Ligamentotaxis can be used for reduction of fragments. Reduction of the shape and height of the calcaneus is easy with the use of gradual distraction. They concluded that rings attached to the distal tibia are not necessary.

Possible complications associated with the external fixator include: uncontrollable edema with drainage exiting at the pin tract sites, pin tract infections, pin loosening, pin irritation, pin/wire breakage, thermal necrosis, non-union, delayed union, malunion, osteomyelitis, joint contractures/subluxation, wound dehiscence, compartment syndrome, reflex sympathetic dystrophy and fracture after frame removal. [2] Many of these complications can be avoided with post-operative compliance and follow-up care. Edema can be alleviated by elevation and partial weight-bearing immediately post-op. Another potential complication is severe pain and damage due to pins or wires compromising muscles, tendons or neurovascular structures.

This complication is decreased with the use of the VCAM™, as no pins are passed through the leg. Major complications such as infection and wire breakage alter the postoperative course and often require removal of the external fixator. [9]

In the cases presented it is evident that the VCAM™ can be constructed in many configurations and therefore be used to treat a variety of lower limb deformities that could lead to ulcerations. In the first case the VCAMTM was applied to achieve gradual correction of a triplanar deformity. It was successful in doing so without the use of leg pins or wires. In the second case it was used a traditional Ilizarov frame to simply offload the extremity to assist in healing two plantar ulcers. In the third case it was again constructed as an Ilizarov type frame to offload a plantar ulcer and to provide compression of the midfoot to the hindfoot. In all three of these cases the VCAM™ was successful and proved to be a useful device to heal ulcerations and correct deformities without the use of leg pins or wires. One of the disadvantages of the VCAM™ as seen in the second case was the development of pin tract infections. This is the most common disadvantage with any external fixator, but the absence of leg pins in our opinion decreases the chance of pin tract infections with the VCAM™. More case studies and research should be explored with the VCAM™ in the areas of trauma especially Lisfranc fractures since it is a midfoot deformity and other lower limb deformities.

In conclusion we feel that the VCAM™ is an excellent modality when managing limb deformities that have lead to the development of ulcerations. It provides a means of realigning the foot in all necessary planes while simultaneously offloading ulcerations. The benefits greatly outweigh the risks associated with use of this device. We have seen good results using this device and recommend it for offloading ulcerations secondary to limb deformities. More case studies and research should be explored with the VCAM™ in the areas of trauma especially Lisfranc fractures since it is a midfoot deformity and other lower limb deformities.

References

1. Hamilton GA, Ford FA. External fixation of the foot and ankle Elective indications and techniques for external fixation in the midfoot. Clin Podiatr Med Surg 2003 20:45-63.
2. Baker MJ, Offutt SM. External fixation Indications and patient selection. Clin Podiatr Med Surg 2003;20:9-26.
3. Vito GR, Talarico LM, Kanuck DM. Use of external fixation to correct deformities of the lower leg. Clin Podiatr Med Surg 2003 20:119-157.
4. Cooper PS. Application of external fixators for management of Charcot deformities of the foot and ankle. Foot Ankle Clin N Am 2002 7:207-254.
5. Cooper PS. Application of external fixators for management of Charcot deformities of the foot and ankle. Semin Vasc Surg 2003 16: 67-78.
6. Jolly GP, Zgonis T, Polyzois V. External fixation in the Management of Charcot Neuroarthropathy. Clin Podiatr Med Surg 2003 20:741-756.
7. Herbst, S. External fixation of Charcot Arthropathy. Foot Ankle Clin N Am 2004 9:595-609.
8. Malizos KN, Bargiotas K, Papatheodorou L, Dimitroulias A, Karachalios T. The below-the-ankle circular frame: A new technique for the treatment of displaced calcaneal fractures. J Foot and Ankle Surg 2005 45(5):295-299.
9. Bevilacqua NJ, Rogers LC. Surgical management of Charcot midfoot deformities. Clin Podiatr Med Surg 200825:81-94.


Send correspondence to: Michael P. DellaCorte, DPM, FACFAS, Director of Podiatric Medical and Surgery Program at NorthShore Forest Hills LIJ; 5901 69th street, Maspeth, NY 11378. (718) 639-3338

1 Director of Podiatric Medical and Surgery Program at NorthShore Forest Hills LIJ; 5901 69th street, Maspeth, NY 11378.
2 PM&S 36 Resident; Hahnemann University Hospital.
3 PM&S 36 Resident; Hahnemann University Hospital.
4 Private Practice New York, NY.

© The Foot and Ankle Online Journal, 2011

May-Hegglin and other Platelet Dysfunctions as Complications to Compartment Syndrome: A case report

by Jason R. Miller, FACFAS, FAPWCA1, Peter Moyer, DPM2

The Foot & Ankle Journal 1 (9): 1

Compartment syndrome is a well known surgical emergency encountered by physicians on trauma call. When compounded by platelet dysfunction, the management of a compartment syndrome becomes exponentially more difficult for the surgeon. The following case describes a twenty-four year old male who sustained multiple comminuted tarsal and metatarsal fractures after a crush injury that was further complicated by an existing platelet dysfunction known as May-Hegglin anomaly (MHA). This article reviews May-Hegglin and other rare hematological conditions that often obscure otherwise straightforward surgical cases.

Key words: May-Hegglin, MHA, compartment syndrome, external fixation, foot fractures

This is an Open Access article distributed under the terms of the Creative Commons Attribution License.  It permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. ©The Foot & Ankle Journal (www.faoj.org)

Accepted: August, 2008
Published: September, 2008

ISSN 1941-6806
doi: 10.3827/faoj.2008.0109.0001

May-Hegglin anomaly (MHA) is a familial disorder that is a rare type of autosomal dominant platelet disorder. From 2000-2005, only 85 families with MHA were reported. [6] It is associated with thrombocytopenia with varying degrees of purpura, bleeding, giant platelets, and cytoplasmic inclusion bodies that resemble Döhle bodies in the granulocytes (neutrophils, eosinophils, monocytes). [4,6] In these patients, neutrophil and platelet function is considered to be normal. Thrombocytopenia occurs in almost all patients and severe bleeding is rare but has been reported.

These patients may have a range of symptoms from asymptomatic to recurrent epistaxis, gingival bleeding, easily bruising to menorrhagia. MHA has not been associated with higher rates of infection. [4]

In 1909, German physician May described a young female patient who had leukocytic inclusions, who was asymptomatic. [6] In 1945, Swiss doctor Hegglin described a father and his two sons who had a triad of thrombocytopenia, giant platelets, and leukocytic inclusions. [6]

These patients have a mutation of the MYH9 gene, encoding non-muscle myosin heavy chain IIA, present in chromosomal region 22q12-13. [4,6] This mutation results in disordered production of non-muscle myosin heavy-chain type IIA.

The result is macrothrombocytopenia, secondary to defective megakaryocyte maturation and fragmentation. Other associated syndromes are Sebastian, Fechtner, or Epstein syndromes. Differential diagnosis associated with thrombocytopenia and large platelets include Alport syndrome, Bernard-Soulier syndrome, Montreal platelet syndrome, immune thrombocytopenia, and gray-platelet syndrome. [6] The differential diagnosis for leukocytic inclusions includes septicemia, myeloproliferative disorders, and pregnancy.

A case report describes a twenty-four year old male who sustained multiple comminuted tarsal and metatarsal fractures after a crush injury that was further complicated by an existing platelet dysfunction known as May-Hegglin anomaly (MHA).

Case Report

A twenty-four year old deaf man was transported from a local hospital to our Level 1 trauma center for evaluation. He was at work when a steel industrial loading dock door came crashing down and landed across his left foot. (Fig. 1)

Figure 1 Initial presentation after crush injury of the left foot. 

Initial evaluation in the trauma bay was significant for left foot swelling, pain, and mottled skin. His sensory function was compromised while gross motor function remained intact. He presented with stable vital signs.

His past medical history was positive for the May-Hegglin anomaly. He reported living with his parents, denied allergies, and had an otherwise unremarkable review of systems. A full physical exam was normal with the exception of his left lower extremity.

The lower extremity exam was positive for: diminished pulses, exquisite pain on palpation of the mid-foot area, pain with range of motion of digits 1,2,3 and 4, decreased temperature, color changes, and swelling. Arterial line pressure monitoring revealed compartment pressures between 75 mmHg and 100 mmHg in the foot, therefore the operating room was called and prepared for emergent surgery.

Plain film and CT scan revealed the following fractures: comminuted intra-articular fracture of the calcaneus, comminuted fractures of the navicular, cuboid, proximal portions of the cuneiforms and fractures at the base of the second and third metatarsals. (Figs. 2,3)

Figure 2 Radiograph reveals first and second metatarsal crush fracture. 

Figure 3  Sagittal CT view of crush injury.  Displaced metatarsal, calcaneal, and cuneiform fractures are evident. 

Stat labs revealed the following abnormalities: WBC 4.9, HgB 8.6, HCT 25.9 and platelets were 39,000mm3. He was then typed and crossed for surgery.

Surgical Procedure

In the operating room, general anesthesia was administered and an emergent fasciotomy was preformed following typical sterile preparation. His left foot was noted to be severely cyanotic, mottled, and cool to touch. An 8-10 cm medial incision was made to the level of the deep fascia.

After the deep fascia was penetrated via blunt dissection, copious amounts of dark, non-coagulated blood flowed from the incision site. (Fig. 4)

Figure 4  Surgical exploration shows dark, non-coagulated blood and hematoma associated with the compartment syndrome. 

Both the medial and plantar compartments were explored through this incision. Approximately one to two minutes after initial incision was made, the hallux changed from a mottled, blanched, cyanotic color to a healthy pink hue with appropriate capillary refill time. A second incision was then made between the shafts of the second and third metatarsals. Blunt dissections in to the deep fascia revealed additional copious amounts of dark blood that was evacuated from the compartment. A third incision was placed between the fourth and fifth metatarsals, and again this compartment was relieved of congestion. Within five minutes after initial incision, the entire foot was pink and warm with a dramatic decrease in the swelling. Further evaluation noted that the rear-foot remained mottled and cyanotic. At that point a fourth incision was made anterior to the Achilles tendon into the deep fascia, and approximately 5 cc of dark blood was evacuated from the calcaneal compartment. The incisions were flushed and packed with saline soaked nu-gauze packing. Attention was then paid to the medial aspect of the calcaneus where a closed reduction of the sustentacular fragment was performed under fluoroscopy.

An external fixator device was placed in triangular fashion under fluoroscopy to maintain proper alignment of the destabilized midfoot and forefoot fractures.

Post-operatively, a posterior splint with a mild compressive dressing was applied and CBC was collected. Medical and hematology consults were activated, neurovascular evaluations were ordered every two hours, cefazolin 1g every 8 hours was started, and repeat radiographs and CT scans were performed.

On post-operative day number one (POD #1), hematology recommended transfusions of both platelets and packed red blood cells prior to the surgical procedure scheduled for POD #5. While they recommend the use of SCD’s, compression stockings, and out of bed to chair three times per day, they discouraged the use of heparin or enoxaparin for DVT prophylaxis. Hematology also recommended that in monitoring the patient for active bleeding, the hemoglobin, hematocrit and platelet count should be drawn every 12 hours and to consider desmopression (DDAVP) if the labs worsened.

On POD #4, he was transfused with four units of platelets, two units of packed red blood cells, and was given prophylactic diphenhydramine.

The patient tolerated the transfusion well with no evidence of reaction. On POD # 5, he was taken back to the operating room for a successful wash out, minor debridement and primary delayed closure. The patient was discharged on POD #6 after two normal CBC evaluations.

His uneventful postoperative course was interrupted on his second office visit when it was noticed that there was some displacement at the comminuted first metatarsal-cuneiform joint. He was taken back to the operating room for a possible fusion or re-manipulation/stabilization procedure. Intra-operatively, the joint was easily manipulated back into place, and small Steinman pins were introduced for stability. Additionally, the sustentacular fragment of the calcaneal fracture was definitively fixated with 4.0mm cannulated screw fixation under fluoroscopy by percutaneous technique. The fixation pins and external fixator were removed six weeks later and he has since returned to regular employment approximately 8 months following this injury. He reports no residual deformity or pain and is able to ambulate freely in regular shoegear. (Fig. 5)

Figure 5 Patient post reduction with functional left foot and no residual pain or deformity.

Discussion

It is important to note that platelets play a central role in normal hemostasis and thrombosis. Platelets originate from pluripotent stem cells that undergo differentiation to the megakaryoblast and then to platelets. Normal platelet counts are between 150,000 to 300,000mm3, with thrombocytopenia being defined as a platelet count less than 100,000mm3. Spontaneous bleeding typically becomes evident after counts drop below 20,000mm3 (spontaneous head bleeds < 5,000mm3). [6]

In the circulating form, platelets appear as a smooth discs enclosed within a plasma membrane. This membrane contains a number of receptor glycoproteins that are responsible for platelet function. Within the platelet are two specific types of granules.

The first, alpha granules contain fibrinogen, fibronectin, factors V and VIII, platelet factor 4 and platelet-derived growth factor and transforming growth factor beta. The second type of granule is for non-metabolic pool adenine nucleotides (ADP & ATP), ionized calcium, histamine, serotonin, and epinephrine.6 When a vessel wall is damaged, platelets undergo three reactions: (1) adhesion and shape change, (2) secretion, and (3) aggregation collectively referred to as platelet activation. [4] (Fig. 6)

From May-Hegglin Anomaly, eMedicine, 2008.

Figure 6 2000x blood smear of a MHA patient demonstrating a typical giant platelet with ill defined granulation.  A normal sized platelet is also seen here.  The cytoplasmic inclusion body represents a Dohle body.

 

A cell blood count is essential in starting a workup in these patients. The platelet count is decreased, usually between 40,000-80,000mm3. The platelets are enlarged up to 15mm3 in diameter, with normal morphology. [4] Evaluation at the electron microscopy level reveals normal cell organelles with an increased amount of disorganized microtubuli.

The Wright-stained peripheral blood smear shows cytoplasmic inclusion bodies, most dominant in the neutrophils, but some are present in the eosinphils, monocytes, and basophils.

The inclusions are up to 5µm in size, they are spindle shaped, pale, blue-staining bodies that consist of ribosomes, endoplasmic reticulum, and microfilaments. [4] The inclusions are similar to Döhle bodies and are found in the periphery of the cytoplasm. [4] Bleeding time is typically prolonged in concordance with the degree of thrombocytopenia.

Since patients with MHA do not have significant bleeding problems, treatment should be based on clinical evaluation, laboratory evaluation and following recommendations from a hematologist pre- and post operatively. Though it is rare for a MHA patient to develop severe bleeding intra- and post operatively, the skilled foot and ankle surgeon should be aware of the risk of bleeding requiring transfusions. [5]

Desmopressin acetate (DDAVP), is a synthetic vasopressin analogue that has been used peri-operatively in patients with MHA. It is an altered form of vasopressin in which deamination of hemicysteine at position 1 and substitution of D-arginine for L-arginine at position 8 has occurred. [2] Desmopressin binds to the V2 receptor in renal collecting ducts, increasing water resorption. It also stimulates release of factor VIII from endothelial cells due to stimulation of the V1a receptor. [2] This change in stereochemistry eliminates vasopressor (V1) receptor agonist activity and enhances the antidiuretic (V2) receptor agonist action and prolongs duration of action from 2-6 hours to 6-24 hours. [2]

Desmopressin stimulates the endothelial release of factor VIII and von Willebrand factor into the plasma (V2 receptor effect).

After a slow infusion of 0.3mcg/kg, plasma concentrations of factor VIII and von Willebrand factor is 2-4x greater. [2]  Although it can be unpredictable, desmopressin has been shown to shorten bleeding time in a variety of platelet dysfunctional diseases.

DDAVP has become the drug of choice for prevention and treatment of bleeding in patients with mild hemophilia A and von Willebrand’s disease because of the increase in factor VIII and von Willebrand factor, but its mechanism in platelet disorders is still one of debate. [5]

Sehbai, et al., reported a case where 34-year old woman with known MHA underwent a craniotomy secondary to an intractable seizure disorder since childhood. [5] After an extensive family history, past medical history of the patient, and extensive workup which included; magnetic resonance imaging (MRI) of the brain, positron emission tomography (PET) scan, and 24 hour video EEG, the woman underwent craniotomy and resection of the temporal lobe foci of seizure activity. She was admitted one day prior to surgery and transfused with 6 units of platelets, and one hour before surgery was given DDAVP. Platelets were on standby if needed intra or post operatively. Her postoperative course was uneventful except for mild hyponatremia secondary to the DDAVP. [5]

Chabane, et al., reported a 24 year old female that was diagnosed with severe thrombocytopenia after giving birth. She was later diagnosed with MHA. She later went on to have a second and third child via cesarean section, and she did not receive platelets for either. The third child was affected by the MHA with a platelet count of 49,000mm3 as well as inclusion bodies on blood smear. [1]

Matzdorff, et al., reported on a patient with Fechtner syndrome that underwent a tonsillectomy and was given DDAVP pre-operatively. [3]

The patient was a 24 year old woman with a past medical history of thrombocytopenia and bruised easily in childhood. She had been diagnosed with Sebastian platelet syndrome, had also noted a impairment with her hearing as well as mild hematuria. After a detailed family history it was noted that some relatives had thrombocytopenia and hearing impairment. At the time, a blood smear was obtained and evaluated with electron-microscope, which confirmed that the inclusions were consistent with Fechtner syndrome. The woman underwent extensive laboratory evaluation: modified Ivy bleeding test, platelet aggregation studies with ADP, collagen, and ristocentin, and standard coagulation test. The patient also had a bone marrow biopsy. The pertinent test in this case was the bleeding test which was greater than thirty minutes, normal being 5-8 minutes. [3] The test was repeated after DDAVP was given, and her bleeding time normalized to 7 minutes 30 seconds, and her von Willebrand factor (her base line was above average) antigen had increased from 150% to 282%. On the day of surgery the women received DDAVP 0.4 µg/kg over 30 minutes 1 hour before the start time, and the surgery went uneventful. [3]

Conclusion

May-Hegglin is a rare platelet disorder associated with macrothrombocytopenia, leukocyte inclusions, deafness and nephritis. Patients may experience easy bruising, recurrent epistaxis, gingival bleeding, menorrhagia, and excessive bleeding associated with surgical procedures. A patient that presents with MHA and an un-witnessed fall should get a CT scan to rule out intracranial hemorrhage and internal bleeding. Patients that present with MHA should be evaluated by a hematologist to recommend DDAVP and platelet transfusions when necessary. In this case, MHA likely played a compounding role in the rapid development of the foot compartment syndrome encountered and could have certainly compounded the post-operative course.

This case demonstrates the need for a multi-disciplinary approach to patients exhibiting May Hegglin anomaly and expeditious surgical intervention when this rare patient population experiences a traumatic event. Additionally, it demonstrates the need to take a thorough history to reveal rare disorders, like this one, in an elective surgery population. A lack of proper treatment in patients with rare platelet disorders can certainly lead to devastating complications. It is our sincere hope that this article will serve to guide the foot and ankle surgeon to appropriately recognize and treat complicating disease processes when they present.

References

1. Chabane H, Gallais Y, Pathier D, Tchernia G, Gaussem P. Delivery management in a woman with thrombocytopenia of the May-Hegglin anomaly type. Eur J Obstet Gynecol and Reproduction Bio 99:124-25, 2001.
2. Mahdy A.M., Webster N.R. Perioperative systemic haemostatic agents. British J Anaesthesia 93(6):842-58, 2004.
3. Matzdorff AC, White JG, Malzahn K, Greinacher A. Perioperative management of a patient with Fechtner syndrome. Ann Hematol 80:436-439, 2001.
4. Noris P, Spedini P, Belletti S, Magrini U, Balduini C. Thrombocytopenia, giant platelets, and leukocyte inclusion bodies (May-Hegglin anomaly): clinical and laboratory findings. Am J Med 104:355-60, 1998.
5. Sehbai A, Abraham J, Brown V. Perioperative management of a patient with May-Hegglin anomaly requiring craniotomy. Am J Hematol 79:303-08, 2005.
6. Shafer FE. May-Hegglin Anomaly. eMed J [online], 2003.


 
Address correspondence to: Jason R. Miller, FACFAS, FAPWCA
Chief, Foot and Ankle Surgery, Pennsylvania Orthopaedic Center
Adjunct Associate Professor, Dept. of Surgery, TUSPM
Office: 215-644-6900 , FAX: 215-644-7160
Email: jrmiller71@pol.net

1Chief, Foot and Ankle Surgery, PA Orthopaedic Center. Adjunct Associate Professor, Dept. of Surgery, TUSPM, Philadelphia, Pa. 19107.
2PGY-4, Foot and Ankle Surgery, Temple University Hospital, Philadelphia, PA, 19140.

© The Foot & Ankle Journal, 2008