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Mycobacterium malmoense Septic Arthritis
Journal of Infection (2003) 46: 60±71 doi:10.1053/jinf.2002.1075
CASE REPORTS
Mycobacterium malmoense Septic Arthritis S. E. Whitehead1, K. D. Allen*1, V. E. Abernethy2, L. Feldberg3 and J. B. Ridyard2 Departments of 1Microbiology, 2Medicine and 3Plastic Surgery, Whiston Hospital, Prescot, Merseyside L35 5DR, UK We report a case of septic arthritis of an interphalangeal joint and osteomyelitis of the phalanx due to Mycobacterium malmoense in a 61-year-old man with a 20 year history of rheumatoid arthritis treated with steroids and azathioprine. This was successfully treated with ethambutol, rifampicin and clarithromycin. To our knowledge this is the only reported case of septic arthritis due to this pathogen which is usually associated with respiratory disease or cervical # 2002 The British Infection Society. Published by Elsevier Science Ltd. All rights reserved. lymphadenitis.
Introduction First described in 1977 in Malmo, Sweden, Mycobacterium malmoense, is usually associated with respiratory disease in adults and cervical lymphadenitis in children. In Sweden, 221 cases were described over a 22-year-period and it was second only to Mycobacterium avium as a cause of atypical mycobacterial infection. In the UK it is the second commonest cause of non-tuberculous lymphadenitis [1]. Uncommon presentations have included disseminated disease in immunocompromised patients [2±4], urinary tract infection [3] and tenosynovitis [5,6]. The environment, e.g., water has been suggested as the principal source of infection. M. malmoense is found in rivers in Sweden [1]. It does not appear to have been associated previously with joint or bone infections.
Case Report A 61-year-old man with a 20 year history of rheumatoid arthritis presented in June 2000 with a discharging sinus on the volar aspect of the left thumb. His arthritis had been stabilised over the preceding 3 years with prednisolone 5 mg once daily and azathioprine 50 mg twice daily. He had been treated between 1993 and 1996 with methotrexate, which had been withdrawn because of resultant neutropenia. He had a history of pneumonia and sepsis complicated by renal failure and emphysema. There was no definite history of tuberculosis but reportedly a shadow had been seen on chest X-ray at the age of 14 or 15. He had never received treatment for tuberculosis. At this presentation a calcified focus was seen in the lower zone of the right lung. He had had a synovectomy of the right hand with excision of the ulna styloid process and repair of extensor tendons of
Please address all correspondence to: K. D. Allen, Department of Microbiology, Whiston Hospital, Dragon Lane, Prescot, Merseyside L35 5DR, UK. Tel.: 44161 430 1834; Fax: 44161 430 1824; E-mail address: [email protected] (K. D. Allen). 0163±4453/03/$35.00
little, ring and middle fingers in 1988. This was followed by a wound infection, which necessitated debridement and cleaning. He had also had bilateral hip replacements in 1991 and a left total knee replacement in 1996. In 1993 he was seen by a dermatologist for slow to heal lesions on the scalp which were diagnosed as solar keratosis and folliculitis. There was no history of trauma or surgery to his left hand or phalanges. The patient's total white cell count was 6.4 109/l with neutrophils of 5.4 109/l. Haemoglobin was 16.0 g/dl and platelet count 204 109/l. His CRP was only slightly raised at 12.0 mg/l in May 2000 and 17.0 mg/l in September 2000. Radiological examination of the left thumb revealed a completely dislocated interphalangeal joint of his left thumb with bony destruction of the area and osteomyelitis of the proximal phalanx. He was treated with 6 weeks of flucloxacillin prior to excision of the proximal and distal phalanx. Biopsy material from the proximal end of the distal phalanx and pus from the bone were sent for culture and histology. The latter revealed granulomatous inflammation with necrotising granulomata incorporating Langhan's giant cells. There were areas of caseous necrosis within the bone. A smear for acid-fast bacilli revealed organisms consistent with mycobacteria. Routine bacterial cultures were negative. The patient was commenced on rifampicin, isoniazid and pyrazinamide. By 20 November 2000 his thumb had completely healed. After 6 weeks' incubation, growth of mycobacteria was seen. This was subsequently identified as M. malmoense sensitive to rifampicin, rifabutin, ethambutol, azithromycin and clarithromycin, and resistant to isoniazid, pyrazinamide, streptomycin and ciprofloxacin. The patient's therapy was changed to ethambutol 800 mg/day, rifampicin 600 mg/day and clarithromycin 250 mg twice daily. The clarithromycin was stopped after 3 months and the ethambutol and rifampicin continued for a total of 2 years.
Discussion As far as we are aware there are no previously reported cases of septic arthritis or osteomyelitis due to M. malmoense. This patient was immunocompromised due to steroids and
# 2002 The British Infection Society. Published by Elsevier Science Ltd. All rights reserved.
Case Reports azathioprine prescribed for several years to treat his rheumatoid arthritis. He also had evidence from at least 7 years previously of poor healing of skin lesions, suggesting a reduced immune response. The source of infection in this case is presumed to be environmental. There was no history of surgery or trauma to the area, but we surmise that infection may have entered through an unnoticed break in the skin and flourished because of the already damaged joint and suppressed immunity, or been deposited following haematogenous spread from a distant focus. Radiologically there was evidence of both septic arthritis and osteomyelitis. Mandal et al. [7] in their review of bone and joint infections, describe the pathogenesis of such infections, whereby bone adjacent to a joint may become infected as the cartilage is destroyed, or alternatively primary involvement of the bone can be complicated by secondary invasion of the joint. Henriques et al. [3] in their report of 221 cases of M. malmoense infections describe a number of patients who had become infected following steroid treatment. A 50-year-old woman treated with steroids and azathioprine developed an abscess on the back and an ulcer behind the left knee, with M. malmoense isolated from both sites. A 71-year-old woman treated with steroids for chronic bronchitis developed pyuria with M. malmoense isolated from the urine. Another patient with hairy cell leukaemia on steroids was infected in several different sites. All three patients responded to antimycobacterial chemotherapy. Prince et al. [5] described a M. malmoense tenosynovitis of the wrist in a patient who had been on daily prednisolone for 10 years. Atypical mycobacterial infections of skin, bone and joints have been associated with steroid use [2,8,9] and should be considered in patients where routine microscopy and culture are negative and there is no response to conventional antibiotics.
61 Acknowledgements
We thank the Public Health Laboratory, Aintree University Hospital and the Northern Regional Centre for Mycobacteriology, Newcastle Public Health Laboratory for identification and sensitivity testing of the isolate.
References 1 Pozniak A, Bull T. Recently recognized mycobacteria of clinical significance. J Infect 1999; 38(3): 157±161. 2 Castor B, Juhlin I, Henriques B. Septic cutaneous lesions caused by Mycobacterium malmoense in a patient with hairy cell leukaemia. Eur J Clin Microbiol Infect Dis 1994; 13: 145±148. 3 Henriques B, Hoffner SE, Petrini B, Juhlin I, Whalen P, Kallenius G. Infection with Mycobacterium malmoense in Sweden: report of 221 cases. Clin Infect Dis 1994; 18: 596±600. 4 Zaugg M, Salfinger M, Opravil M, Luthy R. Extrapulmonary and disseminated infections due to Mycobacterium malmoense: case report and review. Clin Infect Dis 1993; 16: 540±549. 5 Prince H, Ispahani P, Baker M. A Mycobacterium malmoense infection of the hand presenting as carpal tunnel syndrome. J Hand Surg 1988; 13-B: 328±330. 6 Syed AA, O'Flanagan J. Mycobacterium malmoense infection. An unusual cause of wrist swelling. J Hand Surg 1998; 23(6): 811±812. 7 Mandal S, Berendt AR, Peacock SJ. Staphylococcus aureus bone and joint infection. J Infect 2002; 44: 143±151. 8 Zvetina JR, Demos TC, Rubinstein H. Mycobacterium intracellulare infection of the shoulder and spine in a patient with steroid-treated systemic lupus erythematous. Skeletal Radiol 1982; 8(2): 111±113. 9 Ho PL, Ho P, Fung BK, Ip WY, Wong SS. A case of Mycobacterium marinum infection following systemic steroid therapy. Scand J Infect Dis 2001; 33(3): 232±233.
doi:10.1053/jinf.2002.1076
Brevibacterium casei Bacteremia and Line Sepsis in a Patient with AIDS W. M. Janda1, P. Tipirneni2 and R. M. Novak2 1
Division of Clinical Pathology, Department of Pathology and 2Division of Infectious Diseases, Department of Medicine, University of Illinios Medical Center at Chicago, Illinois 60612, USA Brevibacteria are obligately aerobic gram-positive bacilli that are associated with milk products and are also found on human skin. Strains of Brevibacterium casei have been found to correspond to Centers for Disease Control coryneform groups B-1 and B-3 and have been isolated from a variety of human clinical specimens. In this report, we describe a case of B. casei bacteremia and sepsis in a patient with AIDS associated with a contaminated Hickmann catheter and review the microbiology and characteristics of these emerging opportunistic pathogens. # 2002 The British Infection Society. Published by Elsevier Science Ltd. All rights reserved. Please address all correspondence to: W. M. Janda, Clinical Microbiology Laboratory, M/C 750, University of Illinois Medical Center at Chicago, 840 S. Wood Street, Chicago, Illinois 60612, USA. Tel.: 1 312 996 5608; Fax: 1 312 413 0156; E-mail address: [email protected] (W. M. Janda).
CASE REPORTS
Mycobacterium malmoense Septic Arthritis S. E. Whitehead1, K. D. Allen*1, V. E. Abernethy2, L. Feldberg3 and J. B. Ridyard2 Departments of 1Microbiology, 2Medicine and 3Plastic Surgery, Whiston Hospital, Prescot, Merseyside L35 5DR, UK We report a case of septic arthritis of an interphalangeal joint and osteomyelitis of the phalanx due to Mycobacterium malmoense in a 61-year-old man with a 20 year history of rheumatoid arthritis treated with steroids and azathioprine. This was successfully treated with ethambutol, rifampicin and clarithromycin. To our knowledge this is the only reported case of septic arthritis due to this pathogen which is usually associated with respiratory disease or cervical # 2002 The British Infection Society. Published by Elsevier Science Ltd. All rights reserved. lymphadenitis.
Introduction First described in 1977 in Malmo, Sweden, Mycobacterium malmoense, is usually associated with respiratory disease in adults and cervical lymphadenitis in children. In Sweden, 221 cases were described over a 22-year-period and it was second only to Mycobacterium avium as a cause of atypical mycobacterial infection. In the UK it is the second commonest cause of non-tuberculous lymphadenitis [1]. Uncommon presentations have included disseminated disease in immunocompromised patients [2±4], urinary tract infection [3] and tenosynovitis [5,6]. The environment, e.g., water has been suggested as the principal source of infection. M. malmoense is found in rivers in Sweden [1]. It does not appear to have been associated previously with joint or bone infections.
Case Report A 61-year-old man with a 20 year history of rheumatoid arthritis presented in June 2000 with a discharging sinus on the volar aspect of the left thumb. His arthritis had been stabilised over the preceding 3 years with prednisolone 5 mg once daily and azathioprine 50 mg twice daily. He had been treated between 1993 and 1996 with methotrexate, which had been withdrawn because of resultant neutropenia. He had a history of pneumonia and sepsis complicated by renal failure and emphysema. There was no definite history of tuberculosis but reportedly a shadow had been seen on chest X-ray at the age of 14 or 15. He had never received treatment for tuberculosis. At this presentation a calcified focus was seen in the lower zone of the right lung. He had had a synovectomy of the right hand with excision of the ulna styloid process and repair of extensor tendons of
Please address all correspondence to: K. D. Allen, Department of Microbiology, Whiston Hospital, Dragon Lane, Prescot, Merseyside L35 5DR, UK. Tel.: 44161 430 1834; Fax: 44161 430 1824; E-mail address: [email protected] (K. D. Allen). 0163±4453/03/$35.00
little, ring and middle fingers in 1988. This was followed by a wound infection, which necessitated debridement and cleaning. He had also had bilateral hip replacements in 1991 and a left total knee replacement in 1996. In 1993 he was seen by a dermatologist for slow to heal lesions on the scalp which were diagnosed as solar keratosis and folliculitis. There was no history of trauma or surgery to his left hand or phalanges. The patient's total white cell count was 6.4 109/l with neutrophils of 5.4 109/l. Haemoglobin was 16.0 g/dl and platelet count 204 109/l. His CRP was only slightly raised at 12.0 mg/l in May 2000 and 17.0 mg/l in September 2000. Radiological examination of the left thumb revealed a completely dislocated interphalangeal joint of his left thumb with bony destruction of the area and osteomyelitis of the proximal phalanx. He was treated with 6 weeks of flucloxacillin prior to excision of the proximal and distal phalanx. Biopsy material from the proximal end of the distal phalanx and pus from the bone were sent for culture and histology. The latter revealed granulomatous inflammation with necrotising granulomata incorporating Langhan's giant cells. There were areas of caseous necrosis within the bone. A smear for acid-fast bacilli revealed organisms consistent with mycobacteria. Routine bacterial cultures were negative. The patient was commenced on rifampicin, isoniazid and pyrazinamide. By 20 November 2000 his thumb had completely healed. After 6 weeks' incubation, growth of mycobacteria was seen. This was subsequently identified as M. malmoense sensitive to rifampicin, rifabutin, ethambutol, azithromycin and clarithromycin, and resistant to isoniazid, pyrazinamide, streptomycin and ciprofloxacin. The patient's therapy was changed to ethambutol 800 mg/day, rifampicin 600 mg/day and clarithromycin 250 mg twice daily. The clarithromycin was stopped after 3 months and the ethambutol and rifampicin continued for a total of 2 years.
Discussion As far as we are aware there are no previously reported cases of septic arthritis or osteomyelitis due to M. malmoense. This patient was immunocompromised due to steroids and
# 2002 The British Infection Society. Published by Elsevier Science Ltd. All rights reserved.
Case Reports azathioprine prescribed for several years to treat his rheumatoid arthritis. He also had evidence from at least 7 years previously of poor healing of skin lesions, suggesting a reduced immune response. The source of infection in this case is presumed to be environmental. There was no history of surgery or trauma to the area, but we surmise that infection may have entered through an unnoticed break in the skin and flourished because of the already damaged joint and suppressed immunity, or been deposited following haematogenous spread from a distant focus. Radiologically there was evidence of both septic arthritis and osteomyelitis. Mandal et al. [7] in their review of bone and joint infections, describe the pathogenesis of such infections, whereby bone adjacent to a joint may become infected as the cartilage is destroyed, or alternatively primary involvement of the bone can be complicated by secondary invasion of the joint. Henriques et al. [3] in their report of 221 cases of M. malmoense infections describe a number of patients who had become infected following steroid treatment. A 50-year-old woman treated with steroids and azathioprine developed an abscess on the back and an ulcer behind the left knee, with M. malmoense isolated from both sites. A 71-year-old woman treated with steroids for chronic bronchitis developed pyuria with M. malmoense isolated from the urine. Another patient with hairy cell leukaemia on steroids was infected in several different sites. All three patients responded to antimycobacterial chemotherapy. Prince et al. [5] described a M. malmoense tenosynovitis of the wrist in a patient who had been on daily prednisolone for 10 years. Atypical mycobacterial infections of skin, bone and joints have been associated with steroid use [2,8,9] and should be considered in patients where routine microscopy and culture are negative and there is no response to conventional antibiotics.
61 Acknowledgements
We thank the Public Health Laboratory, Aintree University Hospital and the Northern Regional Centre for Mycobacteriology, Newcastle Public Health Laboratory for identification and sensitivity testing of the isolate.
References 1 Pozniak A, Bull T. Recently recognized mycobacteria of clinical significance. J Infect 1999; 38(3): 157±161. 2 Castor B, Juhlin I, Henriques B. Septic cutaneous lesions caused by Mycobacterium malmoense in a patient with hairy cell leukaemia. Eur J Clin Microbiol Infect Dis 1994; 13: 145±148. 3 Henriques B, Hoffner SE, Petrini B, Juhlin I, Whalen P, Kallenius G. Infection with Mycobacterium malmoense in Sweden: report of 221 cases. Clin Infect Dis 1994; 18: 596±600. 4 Zaugg M, Salfinger M, Opravil M, Luthy R. Extrapulmonary and disseminated infections due to Mycobacterium malmoense: case report and review. Clin Infect Dis 1993; 16: 540±549. 5 Prince H, Ispahani P, Baker M. A Mycobacterium malmoense infection of the hand presenting as carpal tunnel syndrome. J Hand Surg 1988; 13-B: 328±330. 6 Syed AA, O'Flanagan J. Mycobacterium malmoense infection. An unusual cause of wrist swelling. J Hand Surg 1998; 23(6): 811±812. 7 Mandal S, Berendt AR, Peacock SJ. Staphylococcus aureus bone and joint infection. J Infect 2002; 44: 143±151. 8 Zvetina JR, Demos TC, Rubinstein H. Mycobacterium intracellulare infection of the shoulder and spine in a patient with steroid-treated systemic lupus erythematous. Skeletal Radiol 1982; 8(2): 111±113. 9 Ho PL, Ho P, Fung BK, Ip WY, Wong SS. A case of Mycobacterium marinum infection following systemic steroid therapy. Scand J Infect Dis 2001; 33(3): 232±233.
doi:10.1053/jinf.2002.1076
Brevibacterium casei Bacteremia and Line Sepsis in a Patient with AIDS W. M. Janda1, P. Tipirneni2 and R. M. Novak2 1
Division of Clinical Pathology, Department of Pathology and 2Division of Infectious Diseases, Department of Medicine, University of Illinios Medical Center at Chicago, Illinois 60612, USA Brevibacteria are obligately aerobic gram-positive bacilli that are associated with milk products and are also found on human skin. Strains of Brevibacterium casei have been found to correspond to Centers for Disease Control coryneform groups B-1 and B-3 and have been isolated from a variety of human clinical specimens. In this report, we describe a case of B. casei bacteremia and sepsis in a patient with AIDS associated with a contaminated Hickmann catheter and review the microbiology and characteristics of these emerging opportunistic pathogens. # 2002 The British Infection Society. Published by Elsevier Science Ltd. All rights reserved. Please address all correspondence to: W. M. Janda, Clinical Microbiology Laboratory, M/C 750, University of Illinois Medical Center at Chicago, 840 S. Wood Street, Chicago, Illinois 60612, USA. Tel.: 1 312 996 5608; Fax: 1 312 413 0156; E-mail address: [email protected] (W. M. Janda).