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Osteomyelitis of the calcaneus due to atypical Mycobacterium
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Foot and Ankle Surgery 15 (2009) 106–108 www.elsevier.com/locate/fas
Case report
Osteomyelitis of the calcaneus due to atypical Mycobacterium John Michelarakis *, Chryssi Varouhaki Orthopaedic Department of Athens’ Childrens’ Hospital ‘‘P & A.Kyriakou’’, Greece
Abstract World wide tuberculous infection of bone remains common but there are few case reports of atypical mycobacteria causing bone infection. A case is reported of recurrent osteomyelitis of the calcaneus due to such an atypical Mycobacterium and associated with interferon-g receptor deficiency (IFN-g) receptor deficiency. # 2008 European Foot and Ankle Society. Published by Elsevier Ltd. All rights reserved. Keywords: Osteomyelitis; Atypical Mycobacterium; Interferon-g receptor deficiency
1. Introduction A case of osteomyelitis of the calcaneus is reported. The infection recurred despite adequate chemotherapy and surgical treatment. Investigation suggested that this was the result of an atypical mycobacterial infection. This is rare but may be managed in a similar fashion to bone infection due to Mycobacterium tuberculosis.
2. Case report A 4-year-old boy presented to with pain and swelling of the left heel, lasting for about 4 weeks after a trivial nonpenetrating injury. He had no systemic symptoms, his temperature was 37 8C and the only significant sign was tenderness over the medial aspect of the left calcaneus. Radiographs were taken (Fig. 1) which showed a lytic lesion. A full blood count showed a mild leukocytosis with a WCC of 13.3, ESR was 45 mm and the CRP was 7 mg/ L. Blood cultures were negative. A bone scan (Fig. 2) showed increased uptake in the area of the left heel. A CT scan was performed, which showed an osteolytic lesion in the left calcaneus measuring 1.7 cm l.9 cm l.7 cm and which eroded the cortex and contained viscous fluid (Fig. 3). He received IV gentamicin and a second* Corresponding author. E-mail address: [email protected] (J. Michelarakis).
generation cephalosporin for 10 days without any signs of improvement. Exploration, debridement and curettage of the lesion was performed under general anesthesia and pus was sent for culture and sensitivity tests. Tissue samples were sent for histology. Cultures were negative for common aerobic and anaerobic bacteria. Histology showed chronic inflammation with formation of granulomata and areas of necrosis with presence of acid-fast bacilli within epithelioid histiocytes and giant cells. The histological picture suggested infection from mycobacteria. The chest X-ray was negative for primary tuberculosis and a negative Mantoux test was obtained. On this basis the bacilli were assumed to be atypical mycobacteria and the patient was started on Clarithromycin, Amikacin and Rifampicin. Immunological tests were performed and initially peripheral lymphocyte phenotype, polymorphonuclear function (NBT test), and IFN-g were all normal. The CRP, ESR and WCC became normal after treatment for 6 months. Follow up radiographs, showed slow but steady improvement and gradual filling of the calcaneal defect. 3 years later, the patient (now aged 6.5 years) presented again with pain in the left heel and a WCC of 12.2 with a predominant lymphocytic type. Radiographs revealed recurrence of the lesion (Fig. 4) and a bone scan showed increased uptake in the area of the left calcaneum. CT scan showed a large osteolytic area within the left calcaneus with erosion and destruction of the medial cortex.
1268-7731/$ – see front matter # 2008 European Foot and Ankle Society. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.fas.2008.07.003
J. Michelarakis, C. Varouhaki / Foot and Ankle Surgery 15 (2009) 106–108
107
Fig. 1. Lateral radiograph showing lytic changes in the calcaneus.
Fig. 3. CT scan showing cystic changes in the calcaneus.
The patient was taken again to theatre for debridement and new pus specimens were sent for culture, including TB, which were all negative. The Mantoux test remained negative. Further immunological test showed interferon-g receptor deficiency but no other abnormality. This was considered to be a recurrence of the atypical mycobacterial infection and the patient was started on Clarithromycin, Rifampicin, Ciprofloxacin and Amicacin. His clinical picture improved gradually and inflammatory markers returned to normal. He remained well 3 years later.
reports of atypical mycobacterial osteomyelitis in immunodeficient patients in the literature, there was no evidence of HIV infection, leukemia or previous immunosuppressive treatment in this case [3,4]. These infections are otherwise almost always associated either with an IFN-g deficiency or more rarely with an IFN-g receptor deficiency, as was the case here [2,5]. In this case it was not possible to identify the precise organism on culture. The diagnosis was based on the typical histology and the persistently negative Mantoux test.
3. Discussion Atypical mycobacterial bone infections are very rare, they are associated with an immunodeficiency and they are very difficult to treat [1,6]. Although there have been case
Fig. 2. Isotope bone scan showing increased uptake in the calcaneus.
Fig. 4. Lateral radiograph showing evidence of recurrent infection in the calcaneus.
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J. Michelarakis, C. Varouhaki / Foot and Ankle Surgery 15 (2009) 106–108
References [1] Hayek S, Issakov J, Ezra E, Wientroub S, Yaniv M. Atypical mycobacterial granulomatous epiphyseal osteomyelitis in an immunocompetent child. J Pediatr Orthop B 2003;12(2 (March)):151–4. [2] Kristjansson M, Arbeit RD. Mycobacterium ulcerans and osteomyelitis. N Engl J Med 1993;329(8):19. 582; (author reply 583).. [3] Redaelli de Zinis LO, Tironi A, Nassif N, Ghizzardi D. Temporal bone infection caused by atypical Mycobacterium: case report and review of the literature. Ontol Neurotol 2003;24(6):843–9.
[4] Lazzarinin L, Mader JT, Calhoun JH. Osteomyelitis in long bones. J Bone Joint Surg (Am) 2004;86:2305–18. [5] Solheim LF, Kjelsberg F. Recurrent mycobacterial osteomyelitis. Report of a case due to Mycobacterium avium–intracellulare–scrofulaceum complex and BCG vaccination. Arch Orthop Trauma Surg 1982;100(4):277–80. [6] Frosch M, Roth J, Ullrich K, Harms E. Successful treatment of Mycobacterium aviumosteomyelitis and arthritis in anon-immunocompromised child. Scan J Infect Dis 2000;32(3):328–9.
Foot and Ankle Surgery 15 (2009) 106–108 www.elsevier.com/locate/fas
Case report
Osteomyelitis of the calcaneus due to atypical Mycobacterium John Michelarakis *, Chryssi Varouhaki Orthopaedic Department of Athens’ Childrens’ Hospital ‘‘P & A.Kyriakou’’, Greece
Abstract World wide tuberculous infection of bone remains common but there are few case reports of atypical mycobacteria causing bone infection. A case is reported of recurrent osteomyelitis of the calcaneus due to such an atypical Mycobacterium and associated with interferon-g receptor deficiency (IFN-g) receptor deficiency. # 2008 European Foot and Ankle Society. Published by Elsevier Ltd. All rights reserved. Keywords: Osteomyelitis; Atypical Mycobacterium; Interferon-g receptor deficiency
1. Introduction A case of osteomyelitis of the calcaneus is reported. The infection recurred despite adequate chemotherapy and surgical treatment. Investigation suggested that this was the result of an atypical mycobacterial infection. This is rare but may be managed in a similar fashion to bone infection due to Mycobacterium tuberculosis.
2. Case report A 4-year-old boy presented to with pain and swelling of the left heel, lasting for about 4 weeks after a trivial nonpenetrating injury. He had no systemic symptoms, his temperature was 37 8C and the only significant sign was tenderness over the medial aspect of the left calcaneus. Radiographs were taken (Fig. 1) which showed a lytic lesion. A full blood count showed a mild leukocytosis with a WCC of 13.3, ESR was 45 mm and the CRP was 7 mg/ L. Blood cultures were negative. A bone scan (Fig. 2) showed increased uptake in the area of the left heel. A CT scan was performed, which showed an osteolytic lesion in the left calcaneus measuring 1.7 cm l.9 cm l.7 cm and which eroded the cortex and contained viscous fluid (Fig. 3). He received IV gentamicin and a second* Corresponding author. E-mail address: [email protected] (J. Michelarakis).
generation cephalosporin for 10 days without any signs of improvement. Exploration, debridement and curettage of the lesion was performed under general anesthesia and pus was sent for culture and sensitivity tests. Tissue samples were sent for histology. Cultures were negative for common aerobic and anaerobic bacteria. Histology showed chronic inflammation with formation of granulomata and areas of necrosis with presence of acid-fast bacilli within epithelioid histiocytes and giant cells. The histological picture suggested infection from mycobacteria. The chest X-ray was negative for primary tuberculosis and a negative Mantoux test was obtained. On this basis the bacilli were assumed to be atypical mycobacteria and the patient was started on Clarithromycin, Amikacin and Rifampicin. Immunological tests were performed and initially peripheral lymphocyte phenotype, polymorphonuclear function (NBT test), and IFN-g were all normal. The CRP, ESR and WCC became normal after treatment for 6 months. Follow up radiographs, showed slow but steady improvement and gradual filling of the calcaneal defect. 3 years later, the patient (now aged 6.5 years) presented again with pain in the left heel and a WCC of 12.2 with a predominant lymphocytic type. Radiographs revealed recurrence of the lesion (Fig. 4) and a bone scan showed increased uptake in the area of the left calcaneum. CT scan showed a large osteolytic area within the left calcaneus with erosion and destruction of the medial cortex.
1268-7731/$ – see front matter # 2008 European Foot and Ankle Society. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.fas.2008.07.003
J. Michelarakis, C. Varouhaki / Foot and Ankle Surgery 15 (2009) 106–108
107
Fig. 1. Lateral radiograph showing lytic changes in the calcaneus.
Fig. 3. CT scan showing cystic changes in the calcaneus.
The patient was taken again to theatre for debridement and new pus specimens were sent for culture, including TB, which were all negative. The Mantoux test remained negative. Further immunological test showed interferon-g receptor deficiency but no other abnormality. This was considered to be a recurrence of the atypical mycobacterial infection and the patient was started on Clarithromycin, Rifampicin, Ciprofloxacin and Amicacin. His clinical picture improved gradually and inflammatory markers returned to normal. He remained well 3 years later.
reports of atypical mycobacterial osteomyelitis in immunodeficient patients in the literature, there was no evidence of HIV infection, leukemia or previous immunosuppressive treatment in this case [3,4]. These infections are otherwise almost always associated either with an IFN-g deficiency or more rarely with an IFN-g receptor deficiency, as was the case here [2,5]. In this case it was not possible to identify the precise organism on culture. The diagnosis was based on the typical histology and the persistently negative Mantoux test.
3. Discussion Atypical mycobacterial bone infections are very rare, they are associated with an immunodeficiency and they are very difficult to treat [1,6]. Although there have been case
Fig. 2. Isotope bone scan showing increased uptake in the calcaneus.
Fig. 4. Lateral radiograph showing evidence of recurrent infection in the calcaneus.
108
J. Michelarakis, C. Varouhaki / Foot and Ankle Surgery 15 (2009) 106–108
References [1] Hayek S, Issakov J, Ezra E, Wientroub S, Yaniv M. Atypical mycobacterial granulomatous epiphyseal osteomyelitis in an immunocompetent child. J Pediatr Orthop B 2003;12(2 (March)):151–4. [2] Kristjansson M, Arbeit RD. Mycobacterium ulcerans and osteomyelitis. N Engl J Med 1993;329(8):19. 582; (author reply 583).. [3] Redaelli de Zinis LO, Tironi A, Nassif N, Ghizzardi D. Temporal bone infection caused by atypical Mycobacterium: case report and review of the literature. Ontol Neurotol 2003;24(6):843–9.
[4] Lazzarinin L, Mader JT, Calhoun JH. Osteomyelitis in long bones. J Bone Joint Surg (Am) 2004;86:2305–18. [5] Solheim LF, Kjelsberg F. Recurrent mycobacterial osteomyelitis. Report of a case due to Mycobacterium avium–intracellulare–scrofulaceum complex and BCG vaccination. Arch Orthop Trauma Surg 1982;100(4):277–80. [6] Frosch M, Roth J, Ullrich K, Harms E. Successful treatment of Mycobacterium aviumosteomyelitis and arthritis in anon-immunocompromised child. Scan J Infect Dis 2000;32(3):328–9.