Orthopedics

Case Reports 

Recurrent Fluctuant Mass of the Wrist and Forearm Associated with Chronic Tenosynovitis by Mycobacterium kansasii

George A. Mazis, MD, MSc; Vasileios I. Sakellariou, PhD, MSc; Fanourios Kontos, MD; Loukia Zerva, MD; Sarantis G. Spyridonos, MD, PhD

Abstract

This article presents a case of a painless fluctuant mass on the volar aspect of the wrist and forearm of an immunocompetent 45-year-old man with no history of significant underlying disease. This mass proved to be a chronic tenosynovitis associated with Mycobacterium kansasii infection. The patient, who had a history of multiple minor cuts and abrasions plus exposure to an aquatic environment, had a wide resection of the lesion and elective tenosynovectomy. Operative findings revealed a marked tenosynovitis of flexor tendons. Several rice bodies lesions were also observed along the course of the involved flexor tendons.

Biopsy showed a granulomatous inflammatory reaction. Specimens of affected tissue were sent to a laboratory for solid (at 30°C and at 37°C) and liquid (at 37°C) mycobacterial culture. The initial Ziehl-Neelsen stain for acid-fast bacilli was positive. After 8 days of incubation, acid-fast bacilli were recovered. In accordance with the diagnosis of M kansasii tenosynovitis and the results of antibiotic susceptibility testing, triple therapy with rifampicin, isoniazid and clarithromycin was initiated. After 3 months of therapy, the patient experienced improvement in the swelling and is due to receive a total of 12 months of antibiotic therapy. Despite awareness of atypical mycobacterial infections, diagnosis is frequently delayed, leading to increased morbidity. Patients with exposure to these atypical pathogens require a broadened differential to include appropriate testing and culture of specimens to obtain an accurate diagnosis.

Chronic tenosynovitis caused by nontuberculous mycobacteria is uncommon in clinical practices. The most common reported species are Mycobacterium marinum and Mycobacterium kansasii, whereas there are reported cases with other species such as Mycobacterium avium complex, Mycobacterium malmoense, Mycobacterium szulgai, Mycobacterium abscessus, Mycobacterium xenopi, and Mycobacterium terrae.1-3

The etiology remains unclear; however, the disease appears to be related to previous trauma, contamination during previous surgical procedures, local corticosteroid injections, or environmental factors (prolonged exposure to water or soil). Immunosuppression is sometimes associated with these infections and can be considered as a risk factor.2 Bernard et al4 described a retrospective study of 10 patients in France and a review of 40 previously published cases that had M kansasii septic arthritis. Twenty-six out of 50 patients had no underlying disease.4 One disease characteristic is that most cases are delayed in diagnosis.

This article presents a case of a painless fluctuant mass on the volar aspect of the wrist and forearm of a 45-year-old man, which was misdiagnosed and left untreated for 3 years and proved to be a chronic tenosynovitis associated with M kansasii infection.

A mariner with no significant underlying disease presented with painless swelling of his left wrist and forearm.

There was no fever, or numbness of the hand, wrist, or forearm. He had a history of multiple minor cuts and abrasions, plus exposure to an aquatic environment due to his occupation. His past history was uneventful and he had no other relevant reports. He was taking no medications and had no history of allergies, smoking, alcohol, or illicit drug use. A review of systems was otherwise unremarkable.

Over 3-year period, he had been treated with multiple aspirations of a gel-like fluid and had received several regimens of nonsteroidal antiinflammatory drugs, but had never been treated by steroid injection.

Clinical examination showed a nontender fluctuant swelling over the palmar aspect of the wrist and forearm and some swelling of the hypothenar eminence (Figure 1).

Laboratory peripheral blood studies showed that complete blood count was within normal range, erythrocyte sedimentation rate was 5 mm/hr, and C-reactive protein was negative. Tuberculin skin testing gave positive results at 19 mm of induration. Liver enzymes and kidney function were normal. Human immunodeficiency virus status, rheumatoid…

Abstract

This article presents a case of a painless fluctuant mass on the volar aspect of the wrist and forearm of an immunocompetent 45-year-old man with no history of significant underlying disease. This mass proved to be a chronic tenosynovitis associated with Mycobacterium kansasii infection. The patient, who had a history of multiple minor cuts and abrasions plus exposure to an aquatic environment, had a wide resection of the lesion and elective tenosynovectomy. Operative findings revealed a marked tenosynovitis of flexor tendons. Several rice bodies lesions were also observed along the course of the involved flexor tendons.

Biopsy showed a granulomatous inflammatory reaction. Specimens of affected tissue were sent to a laboratory for solid (at 30°C and at 37°C) and liquid (at 37°C) mycobacterial culture. The initial Ziehl-Neelsen stain for acid-fast bacilli was positive. After 8 days of incubation, acid-fast bacilli were recovered. In accordance with the diagnosis of M kansasii tenosynovitis and the results of antibiotic susceptibility testing, triple therapy with rifampicin, isoniazid and clarithromycin was initiated. After 3 months of therapy, the patient experienced improvement in the swelling and is due to receive a total of 12 months of antibiotic therapy. Despite awareness of atypical mycobacterial infections, diagnosis is frequently delayed, leading to increased morbidity. Patients with exposure to these atypical pathogens require a broadened differential to include appropriate testing and culture of specimens to obtain an accurate diagnosis.

Chronic tenosynovitis caused by nontuberculous mycobacteria is uncommon in clinical practices. The most common reported species are Mycobacterium marinum and Mycobacterium kansasii, whereas there are reported cases with other species such as Mycobacterium avium complex, Mycobacterium malmoense, Mycobacterium szulgai, Mycobacterium abscessus, Mycobacterium xenopi, and Mycobacterium terrae.1-3

The etiology remains unclear; however, the disease appears to be related to previous trauma, contamination during previous surgical procedures, local corticosteroid injections, or environmental factors (prolonged exposure to water or soil). Immunosuppression is sometimes associated with these infections and can be considered as a risk factor.2 Bernard et al4 described a retrospective study of 10 patients in France and a review of 40 previously published cases that had M kansasii septic arthritis. Twenty-six out of 50 patients had no underlying disease.4 One disease characteristic is that most cases are delayed in diagnosis.

This article presents a case of a painless fluctuant mass on the volar aspect of the wrist and forearm of a 45-year-old man, which was misdiagnosed and left untreated for 3 years and proved to be a chronic tenosynovitis associated with M kansasii infection.

Case Report

A mariner with no significant underlying disease presented with painless swelling of his left wrist and forearm.

There was no fever, or numbness of the hand, wrist, or forearm. He had a history of multiple minor cuts and abrasions, plus exposure to an aquatic environment due to his occupation. His past history was uneventful and he had no other relevant reports. He was taking no medications and had no history of allergies, smoking, alcohol, or illicit drug use. A review of systems was otherwise unremarkable.

Over 3-year period, he had been treated with multiple aspirations of a gel-like fluid and had received several regimens of nonsteroidal antiinflammatory drugs, but had never been treated by steroid injection.

Clinical examination showed a nontender fluctuant swelling over the palmar aspect of the wrist and forearm and some swelling of the hypothenar eminence (Figure 1).

Laboratory peripheral blood studies showed that complete blood count was within normal range, erythrocyte sedimentation rate was 5 mm/hr, and C-reactive protein was negative. Tuberculin skin testing gave positive results at 19 mm of induration. Liver enzymes and kidney function were normal. Human immunodeficiency virus status, rheumatoid factor, and antinuclear antibodies were negative.

Figure 1
Figure 1: Fluctuant swelling over the palmar aspect of the wrist, forearm, and hypothenar eminence.

There were no granulomas or infiltrates on chest radiograph. Radiography of the hand, wrist, and forearm showed extensive soft tissue swelling, whereas magnetic resonance imaging (MRI) revealed marked tenosynovitis of the flexor tendons.

An elective surgical operation for tenosynovectomy was performed. Operative findings revealed a marked tenosynovitis of the flexor tendons. Several rice bodies were observed along the course of the involved flexor tendons.2

Biopsy showed a granulomatous inflammation reaction, and due to clinical suspicion of atypical mycobacterial infection, specimens of affected tissue were sent to a laboratory for mycobacterial culture, as this is the most sensitive way of diagnosing an atypical mycobacteria infection.5

The specimens proceeded according to standard procedures. Two Lowenstein-Jensen slants were used for solid cultures (1 at 30°C and 1 at 37°C) and the MGIT960 automated system (Becton Dickinson, Franklin Lakes, New Jersey) was used for liquid culture at 37°C. The initial Ziehl-Neelsen stain for acid-fast bacilli was positive. After 8 days of incubation, acid-fast bacilli were recovered in the MGIT960 system (Figure 2).

Figure 2
Figure 2: Microphotography of the M kansasii grown in the MGIT960 liquid medium (manipulation 2400X).

 After 17 and 19 days of incubation, acid-fast bacilli were recovered from the Lowenstein-Jensen slants at 30°C and 37°C respectively. The recovered mycobacterium was identified as M kansasii by the combined use of the commercial kits Genotype Mycobacterium CM and AS (Hain, Lifescience, Nehren, Germany) according to the manufacturer’s instructions (Figure 3).

Figure 3
Figure 3: Microphotography of the M kansasii as shown in the specimen after aspiration and the white cells. (manipulation, 2400X).

 For confirmation of identification of this strain, a 439-bp fragment of the 65-kDa heat shock protein (hsp65) gene was amplified using the protocol and the primers Tb11 and TB12 as previously described. The sequence was compared to other published mycobacterial sequences in the GenBank database. It showed 100% similarity (346/346 identities) with the hsp65 sequence of M kansasii strain ATCC 12478. The hsp65 gene sequence had been deposited in GenBank with accession number HM450377 (Figure 4).

Figure 4
Figure 4: Microphotography of the M kansasii as shown after decontamination of the specimen (manipulation, 1200X).

 Susceptibility testing of M kansasii was performed by a modified microdilution method using the commercially available microplates Sensititre SLOMYCOI (Trek Diagnostic Systems, Cleveland, Ohio) according to the manufacturer’s instructions. The antibiotics tested and the minimum inhibiting concentrations obtained (in parentheses) were: amikacin (64 µg/mL), ciprofloxacin (>16 µg/mL), clarithromycin (1 µg/mL), ethambutol (16 µg/mL), ethionamide (0.3 µg/mL), isoniazid (1 µg/ml), linezolid (4 µg/mL), moxifloxacin (4 µg/mL), rifampin (0.5 µg/mL), rifabutin (<0.25 µg/mL), and streptomycin (64 µg/mL).6 The strain was susceptible to clarithromycin, rifampin, rifabutin, and isoniazid, and resistant to ethambutol, amikacin, ciprofloxacin, and streptomycin.

In accordance with the diagnosis of M kansasii tenosynovitis and the results of antibiotic susceptibility testing, a triple therapy with rifampicin, isoniazid, and clarithromycin was initiated. After 3 months of therapy, the patient experienced improvement in the swelling and is due to receive a total of 12 months of antibiotic therapy.

Discussion

This article presented a case of a 45-year-old man with chronic tenosynovitis of his left forearm and wrist, which was shown to be associated with M kansasii infection. However, chronic tenosynovitides are not uncommon; one should always suspect rare causative factors such as Mycobacterium tuberculosis, fungi infections (including Histoplasma capsulatum and Sporothrix schenkii) and nontuberculous mycobacteria, with M kansasii being among the most commonly reported species.3

Patients with chronic tenosynovitis may present with carpal tunnel syndrome. A tendon rupture is a rare complication, however it may occur when the corrected diagnosis and appropriate treatment are delayed. Erythrocyte sedimentation rate and C-reactive protein are normal in most patients. Magnetic resonance imaging is useful for a diagnosis characterized by a marked synovial thickening around the flexor tendons.

A correct diagnosis is usually made late in the course of illness, typically >6 months.7 Rice-body formation can be noted in patients with nontuberculous mycobacteria chronic tenosynovitis, as in our patient. In our patient, it took 3 years to make the definite diagnosis of M kansasii tenosynovitis. A delayed diagnosis of nontuberculous Mycobacteria tenosynovitis is problematic and attributable to a lack of clinical suspicion. Biopsy tissues with staining of acid-fast bacilli, cultures for Mycobacterium using Lowenstein-Jensen with a long incubation of more than 12 weeks, and using 2 temperatures of 30°C and 37°C are the keys for identification of Mycobacterium species. Our patient’s antimycobacterial regimen will be isoniazid, rifampicin, and clarithromycin for 1 year.4 Given the fact that the patient had a surgical intervention that directly decreased the bacterial burden, the course of antimycobacterial regimen may be adequate.

Another problem regarding therapeutic issue is the duration of medical treatment for this organism. M kansasii is a relatively drug-resistant organism, compared to M tuberculosis. However, a specific medical treatment often results in an excellent clinical response but no known exact time to discontinue the drugs.3 Apart from medical treatment, many reports regarded the surgical intervention as an arbitrary choice of treatment of M kansasii tenosynovitis. Although most reports recommended the surgical treatment of this entity, the indication for surgery is usually due to the worsening symptoms of patients. A previous study described a case of M kansasii tendinitis and fasciitis was successfully treated with only antimycobacterial agents for 2 years.8

The combined medical and surgical approach resulted in a positive outcome. Despite awareness of atypical mycobacterial infections, diagnosis is frequently delayed, leading to increased morbidity. Patients with an exposure to these atypical pathogens require a broadened differential to include appropriate testing and appropriate culture of specimens to obtain an accurate diagnosis.

References

  1. Leader M, Revell P, Clarke G. Synovial infection with Mycobacterium kansasii. Ann Rheum Dis. 1984; 43(1):80-82.
  2. Tangkosakul T, Hongmanee P, Santanirand P, et al. Tenosynovitis caused by mycobacterium kansasii: a case report and literature review. J Infect Dis Antimicrob Agents. 2007; 24(3):143-148.
  3. Kang GC, Gan AW, Yam A, Tan AB, Tay SC. Mycobacterium abscessus hand infections in immunocompetent fish handlers: case report. J Hand Surg Am. 2010; 35(7):1142-1145.
  4. Bernard L, Vincent V, Lortholary O, et al. Mycobacterium kansasii septic arthritis: French retrospective study of 5 years and review. Clin Infect Dis. 1999; 29(6):1455-1460.
  5. Lorenz HM, Dalpke AH, Deboben A, et al. Mycobacterium kansasii tenosynovitis in a rheumatoid arthritis patient with long-term therapeutic immunosuppression. Arthritis Rheum. 2008; 59(6):900-903.
  6. da Silva Telles MA, Chimara E, Ferrazoli L, Riley LW. Mycobacterium kansasii: antibiotic susceptibility and PCR-restriction analysis of clinical isolates. J Med Microbiol. 2005; 54(Pt 10):975-979.
  7. Lidar M, Elkayam O, Goodwin D, et al. Protracted Mycobacterium kansasii carpal tunnel syndrome and tenosynovitis. Isr Med Assoc J. 2003; 5(6):453-454.
  8. Parker MD, Irwin RS. Mycobacterium kansasii tendinitis and fasciitis. Report of a case treated successfully with drug therapy alone. J Bone Joint Surg Am. 1975; 57(4):557-559.

Authors

Drs Mazis and Sakellariou are from the First Department of Orthopedic Surgery and Drs Kontos and Zerva are from the Clinical Microbiology Laboratory, University of Athens, Medical School, ATTIKON University General Hospital, Chaidari, and Dr Spyridonos is from the Department of Hand Surgery, Upper Limb, and Microsurgery, KAT General Hospital, Kifissia, Greece.

Drs Mazis, Sakellariou, Kontos, Zerva, and Spyridonos have no relevant financial relationships to disclose.

Correspondence should be addressed to: George A. Mazis, MD, MSc, First Department of Orthopedic Surgery, University of Athens, Medical School, ATTIKON University General Hospital, 1 Rimini St, 12462, Chaidari, Greece (mazis.giorgos@gmail.com).

doi: 10.3928/01477447-20110317-23

10.3928/01477447-20110317-23

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