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FASXXX10.1177/1938640014565050Foot & Ankle SpecialistFoot & Ankle Specialist

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〈 Case Report 〉 Septic Ankle With Purulence Tracking Up the Flexor Hallucis Longus Tendon Sheath Leading to Deep Venous Thrombosis/ Pulmonary Embolism and Compartment Syndrome Abstract: The differential diagnosis for lower extremity swelling and ankle pain is broad and can have overlapping and related diagnoses. If there is concern for more than one diagnosis, the practitioner should perform a thorough physical examination, order the appropriate studies, and perform the correct procedures to completely diagnose and treat the patient. This article presents the case of a 19-year-old male who presented with 5 days of left ankle pain, fevers, and swelling without any known trauma to the area. Physical examination was concerning for a septic ankle joint, cellulitis, deep venous thrombosis, and compartment syndrome. Duplex venous ultrasound confirmed a deep venous thrombosis

Gregory R. Waryasz, MD, Philip McClure, MD, and Bryan G. Vopat, MD

embolism due to the intra-articular in the popliteal vein. Joint aspiration nature of the flexor hallucis longus of the ankle had gross purulence with tendon sheath. the presence of methicillin-resistant Staphylococcus aureus. The patient Level of Evidence: Case report, was taken emergently to the operating Level IV room where he was found to have gross purulence in the deep posterior compartment, medial and lateral soft Compartment syndrome has been tissues of the ankle, and gross purulence found to be a complication of [deep vein in the ankle joint. The deep posterior thrombosis] DVT.” compartment also had significant muscle necrosis and evidence of compartment syndrome. This case Keywords: compartment syndrome; presents the possibility of a septic ankle flexor hallucis longus; septic ankle; leading to compartment syndrome and deep venous thrombosis; pulmonary deep venous thrombosis/pulmonary embolism



DOI: 10.1177/1938640014565050. From the Department of Orthopaedic Surgery, The Warren Alpert Medical School of Brown University and Rhode Island Hospital, Providence, Rhode Island. Address correspondence to: Gregory R. Waryasz, MD, Department of Orthopaedic Surgery, The Warren Alpert Medical School of Brown University and Rhode Island Hospital, 593 Eddy St, Providence, RI 02903, USA; e-mail: [email protected]. For reprints and permissions queries, please visit SAGE’s Web site at http://www.sagepub.com/journalsPermissions.nav. Copyright © 2014 The Author(s)

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Figure 1. Plain radiographs of left leg showing soft tissue edema.

Image A is a AP radiograph of the left ankle. The arrow is pointing to the swollen area over the medial malleolus where there was subcutaneous purulence. Image B is a lateral radiograph of the left ankle.

Case Report A 19-year-old male with a history of depression and polysubstance abuse including cocaine, marijuana, and alcohol presented with 5 days of left ankle pain after previous evaluations by an outside facility where he was diagnosed with an ankle sprain. He was transferred to our facility and was found to be febrile to 103°F, tachycardic into the 140s, but blood pressures were within normal limits. His physical examination was notable for soft tissue swelling, induration, and edema of the left ankle with mild erythema (Figure 1). He had increasing somnolence throughout the exam and appeared toxic. He had full compartments and severe pain in his calf with passive stretch of the great toe. He had tenderness in his calf extending into his popliteal fossa and a positive Homan’s test. His motor/sensory exam was normal. There was limited passive range of motion of the left ankle joint. Echocardiogram did not reveal any valvular vegetations. Laboratory values revealed a white blood cell count of 18.5, C-reactive

protein of 414.78, erythrocyte sedimentation rate of 99, and lactic acid of 0.9 mEq/L. Two blood cultures from admission grew methicillin-resistant Staphylococcus aureus (MRSA). His left tibiotalar joint was aspirated in a sterile fashion and found to be grossly purulent with a gram stain positive for gram positive cocci. His ankle joint was provisionally irrigated with normal saline in the emergency room. The duplex venous ultrasound of the left leg (Figure 2) showed a deep venous thrombosis (DVT) of the popliteal vein initially with partial occlusion, but repeat ultrasound postoperatively showed complete occlusion. Compartment pressure measurements were not performed as he clinically had compartment syndrome. He was taken emergently to the operating room for surgical debridement of his left septic ankle and 4 compartment fasciotomies. Incision was made over the medial aspect of the ankle in the indurated area and there was gross purulence tracking up to the mid-tibia with multiple areas of soft tissue necrosis. A medial ankle

arthrotomy was performed revealing a large amount of gross purulence in the ankle joint. The medial incision for fasciotomy was made and there was a large amount of gross purulence in the deep posterior compartment with muscle bulging and a significant amount of muscle necrosis. There was no purulence in the superficial compartment. The medial incision was extended into the foot to perform a thorough debridement; however, no purulence was identified in the foot. Foot compartments were soft and therefore no fasciotomies were performed. Next the lateral fasciotomy was performed; examination demonstrated no muscle bulging and purulence in the anterior or lateral compartments of the lower leg or the lateral foot. A thorough irrigation and debridement was performed of both the medial and lateral aspects of the leg. Damp to dry dressings were applied to the open wounds and the patient was transferred to the surgical intensive care unit intubated. Damp to dry dressings were chosen to allow for easier monitoring of the wounds and possible infectious spread. On postoperative day (POD) 1, he was extubated. A repeat venous duplex ultrasound of the left leg was performed after surgical debridement and fasciotomies and found to then be fully occluding the popliteal vein. He was anticoagulated with a heparin drip and transitioned to coumadin after his definitive coverage was performed. Intraoperative cultures grew MRSA, and the patient was treated with vancomycin. He was taken back to the operating room on POD 2 and had debridement of the deep posterior compartment muscle that had evidence of necrosis and continued purulence in the deep posterior compartment and tibiotalar joint (Figure 3).Repeat irrigation and debridement continued to be performed every 2 to 3 days. A peripherally inserted central catheter (PICC) line was placed once blood cultures were negative for 48 hours.

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Figure 2. Duplex venous ultrasound of left leg showing DVT of left popliteal vein and computed tomography angiography of chest showing septic pulmonary emboli.

Image A is the duplex venous ultrasound of the left leg with the arrow pointing to the DVT in the left popliteal vein. Image B is the computed tomography angiography of the chest showing filling defects suggesting pulmonary emboli, nonenhancing medial basilar segment of the right lower lobe consistent with infarct, multiple cavitating nodules consistent with septic emboli, and peripheral consolidations also consistent with infection.

He underwent placement of an inferior vena cava filter on POD 5. Shortly after he became increasingly short of breath, tachypneic, and developed an increased oxygen requirement to 40% on face mask. An arterial blood gas was done showing 7.46/43/58/90%. A chest X-ray was done showing multifocal airspace disease; thus, a computed tomography angiography of the chest was performed (Figure 2) showing multiple areas of necrosis and septic pulmonary emboli. The patient’s bacteremia seeded the DVT, which then went to the lungs. A lung bronchoscopy showed MRSA in the lungs as well. He underwent multiple irrigation, debridement, and wound vac placements until all necrotic tissue was removed and the bed of tissue had granulated enough to allow for a skin graft (Figure 4). He had a total of 7 surgeries with the orthopaedic service prior to his definitive coverage with the plastic surgery service. During the debridements, there was evidence of necrosis of the deep posterior compartment in which all necrotic

tissue was debrided except for the flexor hallucis longus, which continued to be viable. His medial and lateral incisions were unable to be completely closed and required a split thickness skin graft with donor site from the left thigh. He was discharged with a PICC line and treated with intravenous vancomycin for 6 weeks. His skin grafts went on to heal and he has done well with a slight limp with an ankle brace, but no neurovascular sequelae 3 months after initial presentation (Figure 5).

Discussion To our knowledge and extensive literature search, this is the first case reported of a septic ankle arthritis with concomitant compartment syndrome and DVT. A case of septic knee induced compartment syndrome was reported in 2010 that developed acute pulmonary emboli.1 Compartment syndrome has been found to be a complication of DVT in many case reports.2 DVT has also been found to be a complication of

osteomyelitis.3 Knee septic arthritis has also been reported to be associated with acute DVT.4 The concept of material from a septic arthritis from the ankle that can track up the flexor hallucis longus tendon sheath is derived from the intraarticular anatomy of the flexor hallucis longus in some patients. A cadaveric study by Draeger et al5 utilized sequential 2-cc radiopaque dye under fluoroscopy to determine that maximum volume in the ankle joint. Nine cadaver ankles were tested. The mean maximum ankle joint volume was 20.9 ± 4.9 cc (range = 16-30 cc). The mean ankle joint volume at maximum volume was 142.2 ± 13.8 mm Hg (range = 122-166 mm Hg). Two of the 9 specimens showed evidence of fluid tracking into the flexor hallucis longus tendon sheath.5 Elevated pressures in the ankle joint can lead to a compartment syndrome by the communication of the plantarflexion tendon sheaths with the ankle joint.5 Our case may suggest that this communication can also lead to a DVT

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Figure 3. Intraoperative photos from second debridement procedure showing a necrotic deep posterior compartment.

Image A shows the anterior and lateral compartments with good muscle viability. Image B is a close-up image of the medial leg with the arrow pointing to the necrotic muscle in the deep posterior compartment. Image C shows the medial leg and foot.

as well. Most likely in our case, the ankle joint capsule ruptured leading to subcutaneous abscess formation around the ankle joint in addition to the septic joint and collection in the deep posterior compartment. The deep posterior compartment collection may have caused external pressure on the vein resulting in the DVT. The incidence of DVT in children is rare with an incidence less than 0.01%.6 The septic patient is more at risk for DVT given dehydration, inflammation, complement activation, and bedrest.6 DVT also seems to be more common in staphylococcal infections.3,6-8 A case

series of 9 patients at Texas Children’s Hospital was reported showing that patients with acute Staphylococcus aureus osteomyelitis have a unique propensity to developing an adjacent DVT and possibly develop septic emboli.3 More than 75% of the cases reported in the Texas Children’s Hospital series had MRSA.3 Staphylococcus aureus is already known to cause sepsis syndrome, purpura fulminans, necrotizing fasciitis, and severe necrotizing pneumonia.3 MRSA more commonly causes DVT than methicillin-susceptible Staphylococcus aureus.8

Pyomyositis has been reported to be a rare cause of acute compartment syndrome and usually occurs with the causative bacteria being Staphylococcus aureus.9 Pyomyositis is a primary acute bacterial infection of the skeletal muscle that leads to multiple abscess formation. Up to 50% of the cases are not associated with superficial injury including external skin puncture, foreign bodies, or hemaoma.10 Most cases do not have any cellulitis, fasciitis, or periosteal reaction. The cause of the infection in this case was not conclusive; however, this demonstrates an important diagnosis

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Figure 4. Intraoperative photos showing progression from wound vac to final soft tissue coverage.

Image A shows a wound vac applied after a debridement to the lateral leg. Image B shows a partial closure and then wound vac application to the medial ankle due to skin loss. Image C shows the skin graft healing over the medial ankle.

for clinicians to consider. The presence of an MRSA infection that includes septic ankle may coincide with compartment syndrome and a DVT/ pulmonary embolism.

Conclusion The flexor hallucis longus can be an intra-articular structure, and therefore, when pressure in the tibiotalar joint rises, there can be synovial fluid and purulence that can track into the deep posterior compartment. In our case,

the etiology of the septic tibiotalar joint is unclear. The patient did not admit to injecting intravenous drugs. Our hypothesis is that the infection either started as bacteremia and seeded the joint or started in the joint by direct inoculation and over the course of 5 days, purulence went up the flexor hallucis longus tendon sheath into the deep posterior compartment resulting in external pressure on the popliteal vein causing a DVT and the increased contents of the deep posterior compartment also

caused compartment syndrome. The DVT was likely seeded from bacteremia and then became a septic pulmonary embolism. The abscess collections along the medial and lateral ankle were most likely the result of a ruptured ankle joint capsule as there was well over 30 cc collected intraoperatively from the ankle joint and surrounding purulent collections along the medial and lateral ankle. This case demonstrates the need to keep multiple diagnoses in mind when a patient has a complex presentation.

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Figure 5. Three-month follow-up visit.

Image A shows the lateral aspect of the left leg showing the skin graft. Images B and C show the medial aspect of the left leg skin graft areas and areas with granulation tissue.

Authors’ Note Gregory R. Waryasz, MD, and Philip McClure, MD, have no conflicts of interest. Bryan G. Vopat, MD, is a consultant for Dupuy/Mitek.

References 1. Backes J, Taylor BC, Clayton MD. Septic knee-induced deep venous thrombosis in a young adult. Orthopedics. 2010;33:770. doi:10.3928/01477447-20100826-28. 2. Lamborn DR, Schranz C. Compartment syndrome as a complication of ileofemoral deep venous thrombosis: a case presentation. Am J Emerg Med. 2014;32:192. e1-192.e2. doi:10.1016/j.ajem.2013.08.050. 3. Gonzalez BE, Teruya J, Mahoney DH Jr, et al. Venous thrombosis associated with staphylococcal osteomyelitis in

Surg Am. 2007;89:1517-1523. doi:10.2106/ JBJS.F.01102.

children. Pediatrics. 2006;117:1673-1679. doi:10.1542/peds.2005-2009. 4. Rafailidis PI, Kapaskelis A, Falagas ME. Knee septic arthritis due to Streptococcus pyogenes associated with acute thrombosis of the tibial and peroneal veins: case report and review of the literature. Scand J Infect Dis. 2007;39:368-370. doi:10.1080/00365540600978963.

8. Mantadakis E, Plessa E, Vouloumanou EK, Michailidis L, Chatzimichael A, Falagas ME. Deep venous thrombosis in children with musculoskeletal infections: the clinical evidence. Int J Infect Dis. 2012;16:e236-e243. doi:10.1016/j. ijid.2011.12.012.

5. Draeger RW, Singh B, Parekh SG. Quantifying normal ankle joint volume: an anatomic study. Indian J Orthop. 2009;43:72-75. doi:10.4103/00195413.45326.

9. Shedek BK, Nilles EJ. Communityassociated methicillin-resistant Staphylococcus aureus pyomyositis complicated by compartment syndrome in an immunocompetent young woman. Am J Emerg Med. 2008;26:737.e3-4. doi:10.1016/j. ajem.2007.11.034.

6. Walsh S, Phillips F. Deep vein thrombosis associated with pediatric musculoskeletal sepsis. J Pediatr Orthop. 2002;22:329-332. 7. Hollmig ST, Copley LAB, Browne RH, Grande LM, Wilson PL. Deep venous thrombosis associated with osteomyelitis in children. J Bone Joint

10. Harrington P, Scott B, Chetcuti P. Multifocal streptococcal pyomyositis complicated by acute compartment syndrome: case report. J Pediatr Orthop B. 2001;10:120-122.

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pulmonary embolism and compartment syndrome.

The differential diagnosis for lower extremity swelling and ankle pain is broad and can have overlapping and related diagnoses. If there is concern fo...
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