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International Journal of Infectious Diseases (2009) 13, e145—e147

http://intl.elsevierhealth.com/journals/ijid

CASE REPORT caused by Mycobacterium terrae in a patient with

Huan-Wen Chen a, Chih-Cheng Lai b, Che-Kim Tan c,* a Department of Internal , Lotung Poh-Ai Hospital, Yi-Lan, Taiwan b Department of , Yi-Min Hospital, Taipei, Taiwan c Department of , Chi-Mei Medical Center, Yungkang, Tainan, Taiwan and Department of Internal Medicine, Taipei Medical University, Taipei, Taiwan

Received 5 May 2008; received in revised form 23 August 2008; accepted 2 September 2008 Corresponding Editor: Sheldon T. Brown

KEYWORDS Summary To date, few cases of human joint caused by the Mycobacterium terrae Mycobacterium terrae; complex have been reported. Because M. terrae infection is a relatively uncommon problem, it Arthritis can be mistaken for a noninfectious inflammatory joint condition. The most common presentation of M. terrae complex infection is tenosynovitis of the hand; in bones other than those of the hands are rarely reported. Here, we describe a patient with arthritis of the knee caused by M. terrae and review data from other cases reported in the medical literature. # 2008 International Society for Infectious Diseases. Published by Elsevier Ltd. All rights reserved.

Introduction We describe here a patient with arthritis of the knee caused by M. terrae and review data from other cases reported in The Mycobacterium terrae complex includes M. triviale, M. the medical literature. nonchromogenicum and M. terrae.MembersoftheM. ter- rae complex are Runyon group III, non-pigmented, slow- Case report growingorganisms.DespitethecommonopinionthatM. terrae complex isolates are nonpathogenic,1 these organ- The patient was a 31-year-old, HIV-seronegative man with a isms are occasionally identified in clinical diseases of joints, history of methotrexate and systemic steroid treatment for tendons, lung, gastrointestinal tract and genitourinary rheumatoid arthritis. In August 2005, he complained of pain tract. The upper extremities are the most frequently and stiffness in his right knee for about one week. He did not infected sites of the musculoskeletal system, but infections recall any distinct trauma before the onset of his initial joint in bones other than those of the hands are rarely reported..2 lesions. He did not have fever, fatigue, anorexia, regional adenopathy, respiratory or gastrointestinal symptoms, or * Corresponding author. Departments of Intensive Care Medicine, weight changes during the course of this illness. Physical Chi-Mei Medical Center, Yungkang, Tainan, Taiwan. examination was unremarkable except for swelling, local Tel.: +886 6 2812811; fax: +886 6 2828928. heat and tenderness over the right knee joint. E-mail address: [email protected] (C.-K. Tan). was palpated around the right knee. Movements of the right

1201-9712/$36.00 # 2008 International Society for Infectious Diseases. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.ijid.2008.09.002 e146 H.-W. Chen et al.

Table 1 Characteristics of patients with infections in bones other than those of the hand due to Mycobacterium terrae complex.

Case Age No. of months with Comorbid Infected Corticosteroid Outcome (Reference) (years); sex symptoms before condition(s) site(s) diagnosis 1 (5) 0.5; M 0.75 None Hip None Cured 2 (6) 65; M 72 Not specified Ankle Injected Cured 3 (7) 57; M 3 Rheumatoid Knee Injected Improved arthritis 4 (8) 22; unknown 39 None Knee Injected Unknown 5 (present report) 31; M 3.5 Rheumatoid Knee Systemic Improved arthritis

knee were moderately limited in all directions. Laboratory described as an occasionally isolated nonpathogenic colonizer. examination revealed elevated erythrocyte sedimentation To date, few cases of human joint infection caused by the M. rate (ESR: 57 mm/h) and rheumatic factor (460 IU/ml); renal terrae complex have been reported. Because M. terrae infec- and liver function tests, the level of uric acid and blood tion is a relatively uncommon problem, it can be mistaken for a counts were in the normal range. noninfectious inflammatory joint condition. The most common Aspiration of joint effusion yielded 30 ml of serosangui- presentation of M. terrae complex infection is tenosynovitis of nous fluid, but no crystals were found upon microscopic the hand. In most patients with infections of the upper extre- examination. Gram stain and acid-fast stain were negative, mity, there was either a history of an antecedent puncture of as were routine bacterial cultures. Analysis of the synovial the hand or an occupation or hobby that could have increased fluid revealed a white blood cell count of 9450/mm3, with the risk of hand trauma and wound contamination.2 Persistent predominance of neutrophil (78%), total protein of 4.5 gm/ or recurrent infection was reported in 21% of patients, and 10% dl, LDH of 333 IU/l and glucose of 116 mg/dl. Aspirate was eventually underwent amputation.2 inoculated onto Middlebrook 7H11 selective agar with anti- The infection of bones in the hip,5 ankle6 and knee7,8 was microbials (BBL, Becton Dickinson, Sparks, MD, USA) and rarely reported in the English literature. Details from four BACTEC MGIT 960 system bottle (Becton-Dickinson Diagnostic cases and the case reported here are shown in Table 1. The Instrument Systems, Sparks, MD, USA). Acid-fast bacilli (AFB) age of the patients ranged from 6 months to 65 years (median stains were done on submission of the specimens and age, 31 years). Most patients had no predisposing condition repeated on positive cultures from a BACTEC system. Isolates and were thought to be immunologically intact. Only two were identified to the species level using conventional bio- patients had underlying disorders of rheumatoid arthritis. chemical methods.3 A purified protein derivative (PPD) Most of them had improvement or cure after treatment.. In tuberculosis skin test remained negative. After two months contrast to tenosynovitis of the hand, there was neither of inoculation, the mycobacteria culture of synovial fluid apparent exposure history nor associated occupations or yielded a positive result. Antimicrobial therapy with rifatar hobbies (except one farmer). was given for possible tuberculous arthritis. However, the The interval between the onset of symptoms and the clinical condition did not improve and the patient developed identification of the etiologic agent was 23.7 Æ 31.4 months a left knee effusion. The analysis of the fluid was similar to (the mean time Æ SD). Four of these five cases were treated the findings for the right knee. Three weeks later, it was with corticosteroid, either injected locally or given systemi- recognized that the first synovial fluid cultures had been cally, before the diagnosis of infection was made. The long positive for M. terrae. The isolated organism grew at 37 8C interval between the onset of symptoms and the microbio- and showed nonpigmentation in both light and dark condi- logical identification of M. terrae, and the initial use of tions. There was no growth in 5% sodium chloride and on corticosteroid therapy, suggests that the clinical presenta- MacConkey agar. There was a negative 3-day and positive 14- tion was often nonspecific and a noninfectious inflammatory day arylsulfatase test. It was negative for both urease and condition was suspected in most cases. The morbidity asso- pyrazinamidase, but positive for b-galactosidase, acid phos- ciated with delayed diagnosis and the possible adverse phatase, catalase (45 mm foam), thermostable catalase and effects of corticosteroid therapy in untreated chronic infec- Tween 80 hydrolysis (10 days). The patient was then treated tions underscore the importance of including M. terrae with azithromycin (250 mg/daily), ethambutol (800 mg/ infection (and other occult infections) in the differential daily) and levofloxacin (750 mg/daily). Two months later, diagnosis of refractory arthritis that is thought to be of a the swelling and pain in the bilateral knee gradually nonspecific inflammatory nature. improved during follow-up in outpatient clinics. Finally, he Our patient received macrolide and ethambutol for M. was successfully treated with the combined regimen for 12 terrae arthritis and had clinical improvement without any months. sequelae. This is consistent with previous findings that ethambutol and azithromycin are effective in vitro against Discussion the M. terrae isolate tested.2 However, in vitro antibiotic susceptibility testing for this mycobacterial isolate might be M. terrae was first isolated by Richmond and Cummings in worthwhile to refine the therapy, which might have to be 1950 from radish washings.4 This organism is usually continued for a prolonged period to increase the possibility of Mycobacterium terrae arthritis e147 a cure and decrease the need for surgical intervention in such 2. Smith DS, Lindholm-Levy P, Huitt GA, Heifets LB, Cook JL. a localized disease. Mycobacterium terrae: case reports, literature review, and in Our case is instructive for three reasons. First, in spite of vitro antibiotic susceptibility testing. Clin Infect Dis 2000;30: the rare occurrence of M. terrae arthritis of the lower 444—53. 3. Metchock B, Nolte FS, Wallace Jr RJ. Mycobacterium. In: Murray extremities, M. terrae should be considered as one possible PR, Baron EJ, Pfaller MA, Tenover FC, Yolken RH, editors. Manual pathogen of joint infection, in either immunocompromised or of Clinical Microbiology. 7th edition. Washington, DC: ASM Press; immunocompetent patients. Second, entertaining the pos- 1999. p. 399—437. sibility of an indolent mycobacterial infection in such cases 4. Richmond L, Cummings MM. An evaluation of methods of testing might reduce the inappropriate use of corticosteroid therapy the virulence of acid-fast bacilli. Am Rev Tuberc 1950;62: in the context of unsuspected infection. Finally, a multidrug 632—7. antibiotic regimen that includes a macrolide might be useful 5. Dechairo DC, Kittredge D, Meyers A, Corrales J. Septic arthritis in the management of such patients. due to Mycobacterium triviale. Am Rev Respir Dis 1973;108: Conflict of interest: No conflict of interest to declare. 1224—6. 6. Hirata S, Tomiyama T. The right foot joint infection with Myco- bacterium nonchromogenicum. Kekkaku 1976;51:389—92. References 7. Dijkmans BAC, Mouton RP,Macfarlane JD, et al. Bacterial arthritis caused by Mycobacterium terrae. Infection 1981;9:204—7. 1. Wayne LG, Sramek HA. Agents of newly recognized or infrequently 8. Fournie B, Dabernat H, Vaudet B, Jalby H, Fournie A. Arthritis of encountered mycobacterial diseases. Clin Microbiol Rev the knee caused by Mycobacterium terrae. Rev Rheum Mal 1992;5:1—25. Osteoartic 1987;54:49—51.