British Journal of Rheumatology 1990;29:150-153 CASE REPORT
MYCOBACTERIUM
KANSASII INFECTION IN THE WRIST AND HAND
BY J. DILLON, C. MILLSON AND I. MORRIS Department of Rheumatology, Kettering General Hospital, Kettering, Northamptonshire NN168UZ SUMMARY Mycobacterium kansasii infection of the deep structures of the wrist and hand can cause progressive damage which may eventually lead to permanent loss of hand function. This report describes three cases followed by a review of the literature. The important principles of management in this unusual infection are discussed. KEY WORDS:
Tendinitis, Infection, Mycobacterium kansasii.
A 54-year-old man presented in March 1982 with a painful swollen right hand. Two months previously a piece of metal hacksaw blade had entered the dorsum of his right hand. He underwent incision and drainage. Although the hand settled initially, he subsequently complained of pain in the palm and tenderness in the flexor sheaths of the little and index fingers. His past medical history was unremarkable. FBC, ESR and radiographs of the hand and chest were normal. At exploration, the flexor tendons were exposed and the synovial tissue appeared 'swollen and greyish'. Routine microbiological culture of biopsies were negative. However, histology revealed 'non-caseating granulomata consistent with sarcoid'. Ziehl-Neelsen staining was not performed. Over the next few weeks the wrist did not settle in spite of a trial of oral prednisolone for 6 weeks. He presented again with paraesthesiae in the thumb, index and middle fingers associated with pain in his forearm that woke him at night. This was diagnosed as median nerve compression and he was admitted for a release operation. Further tissue was excised, and again histological examination was consistent with sarcoid. In the next 18 months he returned several times with wrist and hand pain and underwent two further exploratory/ debridement procedures. He was referred to the Rheumatology Department where he complained of pain once again, and was found to have a swollen wrist with reduced movement and a weak grip. FBC, ESR and chest X-ray (CXR) were normal but X-ray of the right hand revealed erosions in the radiocarpal and ulnocarpal joints (Fig. 2). A further
CASE REPORTS Case 1 A 42-year-old tube-worker at British Steel presented to the Orthopaedic Department in April 1978 with a 12-month history of swelling of the palmar aspect of the left hand (Fig. 1). The swelling had started in the left middle finger and had spread along the flexor tendon into the palm. Through his work he had been exposed to titanium and nickel but not beryllium. His past medical history was unremarkable and he was otherwise fit and well. On examination there was marked swelling of the left middlefingerextending from the proximal interphalangeal joint down into the palm. There was associated loss of flexion, but no other abnormality. A clinical diagnosis of tendinitis with swelling was made. Full blood count (FBC), erythrocyte sedimentation rate (ESR) and radiographs of the hand and chest were all normal. At exploration, the flexor tendon sheath was found to be inflamed and a biopsy taken, along with fluid for ordinary bacterial culture. The fluid culture was negative but the biopsy was reported as showing non-caseating granulomas with fresh fibrin, consistent with sarcoid. Ziehl—Neelsen stains were not performed at this stage. When reviewed in out-patients some weeks later, the pain had not settled and he was therefore treated with an intrasynovial injection of hydrocortisone acetate and referred to the Rheumatology Department. Similar symptoms and signs as before were found but on this occasion a small amount of fluid was aspirated which subsequently grew Mycobacterium kansasii with the following sensitivities: resistant to para-aminosalicylic acid (PAS) and isoniazid, ethionamide, ethambutol, thiocetazone and cycloserine. He was started on rifampicin and ethambutol but the hand did not respond adequately and he was referred back to the surgeons for a reexploration. When exposed, the flexor tendons were found to be extensively inflamed and necrosed with an associated purulent exudate. The samples grew Mycobacterium kansasii only. He remained on antituberculous chemotherapy for 18 months and was eventually discharged from the clinic with only a minor degree of residual disability. Submitted 14 November 1988; re-vised version accepted 11 August 1989. Correspondence to Dr I. Morris.
FIG. 1.—Case 1. Hand at presentation showing palmar swelling at base of middle finger. 150
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Case 2
THERE are 24 cases of Mycobacterium kansasii synovial infection in the literature and we describe a further three patients with involvement of flexor tendon sheaths.
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M. KAN SASH IN WRIST AND HAND
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on Lowenstein-Jensen media, Mycobacterium kansasii sensitive to rifampicin, ethambutol, ethionamide, capreomycin and cycloserine, resistant to streptomycin and highly resistant to isoniazid. He was given a 12-month course of rifampicin and ethambutol and the pain settled with a virtually full range of movement.
biopsy was undertaken and a specimen sent for histology, Ziehl-Neelsen staining and mycobacterial culture. This grew Mycobacterium kansasii with the following sensitivities: rifampicin, ethionamide, ethambutol, thiocetazone and cycloserine, and resistant to PAS and isoniazid. He was commenced on ethambutol and rifampicin but the rifampicin made him nauseated and dizzy so prothionamide was substituted after a month's course was completed. An 18month course of treatment was given. At his most recent follow-up, although wristflexionand extension were limited, he had little pain and there was good bony fusion on X-ray. Case 3 A 70-year-old retired engineer presented to the Rheumatology Department in July 1983 with a 3-month history of pain in his right hand, particularly noticed when using spanners. The pain was worse in the thumb and index finger and occasionally woke him at night. On examination he had inflammatory synovitis affecting the flexor tendons at the wrist as well as a ganglion on the right wrist. The grip was weaker on the right. A clinical diagnosis of flexor tendinitis and carpal tunnel syndrome was made. Investigations revealed a normal FBC, ESR, RoseWaaler, immunoelectrophoresis, liver function tests and negative autoantibody screen. X-ray of the wrist revealed ectopic calcification, osteoporosis and soft tissue thickening. CXR showed cardiomegaly only. Mantoux test at 1:10000 and Kveim test were both negative. Nerve conduction studies and electromyography showed severe median nerve compression and so he was admitted for exploration and biopsy. At operation the flexor tendon sheaths were found to be thickened and inflamed. These were partially excised and the carpal tunnel decompressed. Histology showed a granulomatous synovitis (Fig. 3). Ziehl-Neelsen stain was negative. However, fluid aspirated from the joint subsequently grew,
*: ^"
HG. 3.—Granuioma from (Jase 3.
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FIG. 2.—Case 2. X-ray of wrist showing loss of joint space in the intercarpal and radiocarpal joints. The joint margins have become indistinct and there is patchy osteoporosis. In addition, the proximal pole of the scaphoid has eroded in through the distal radial cartilage into the metaphysis. Other erosions are present with some proliferative bone repair.
DISCUSSION Details of all recorded synovial infection with Mycobacterium kansasii are outlined in Table I, including our cases. Improvement in morphological characterization may explain a higher rate in more recent years. The tendon sheath of the wrist and hand appears to be the most common site of synovial infection by nontuberculous Mycobacterium in humans [1]. Various aetiological factors have been suggested including injury [1-3], steroid injection [2-5] and a joint damaged by an arthropathy [2, 6,7]. Mycobacterium kansasiihas been found in cows' milk [8] and tap water but personto-person spread does not occur [9]. One of our cases sustained a penetrating injury (case 2) whilst the other two were employed in a job (case 1) or pastime (case 3) that involved hard manual work. Few of the occupations or hobbies of patients described previously were delineated so their significance is purely anecdotal [2, 7,10,11], but it may be that heavy manual labour does expose to the risk of penetrating injuries and thus introduction of the organism. Perhaps the most alarming feature is the diagnostic delay in a few cases [12,13]. In two of the cases we describe, the correct diagnosis was not considered until several months after presentation. In the third case, an atypical Mycobacterium was suspected but the synovial biopsy failed to grow anything and the bacteria was only isolated at joint aspiration some 5 weeks later, when the wrist had failed to settle.
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TABLE I SUMMARY OF CASES SO FAR DESCRIBED INCLUDING OUR OWN*
Presenting feature
Specimen for diagnosis
52 (F)
Flexor tenosynovitis
Synovectomy tissue Known pharyngeal Mycobacterium
43 (F)
Tennis elbow
Synovial culture
61 (M)
Carpal tunnel syndrome
82 (F)
Sudden swelling of joint
Joint aspirate after carpal tunnel release Joint aspirate
47 (M)
Carpal tunnel syndrome Flexor tenosynovitis
Joint aspirate; synovial culture Synovium
Age
Case Ref. 1
[1]
(years)
Time to diagnosis
Treatment
Outcome
3 months
Surgery
Complete recovery
Steroid injection; trauma to elbow
23 months
Surgery and chemotherapy
Pain free. Some residual loss of
Steroid injection
1 month
Surgery, chemotherapy, erythromycin Surgery and chemotherapy
Full function returned
Surgery and chemotherapy Surgery and chemotherapy
Full recovery
Risk factors
Icnrjcncii tVM'lOUijff
2
i n ft i c\ n llllll'llUll
TI
3
[4]
4
[14]
6
44 (F)
7
71 (M)
Flexor tenosynovitis
Synovial biopsy
Steroid injection
4 months
Transient arthropathy; poststeroid injection
6 months
Chemotherapy alone
Nil
18 months
Symptom free
Needs reconstructive surgery to tendon Full recovery
8
*
42 (M)
Flexor tendinitis
Synovial biopsy
9
*
54 (M)
Flexor tendinitis
Synovial biopsy
Penetrating injury
36 months
Surgery and chemotherapy Chemotherapy and surgery
10
*
70 (M)
Joint aspirate
Nil
2 months
Surgery and chemotherapy
11
[7]
68 (F)
Synovial aspiration
Rheumatoid arthritis; azathioprine; prednisolone
8 months
Surgery and chemotherapy
Pain free. Good range of movements
12
[11]
41 (M)
Flexor tendinitis and carpal tunnel syndrome Worsening rheumatoid arthritis; progressive synovitis Carpal tunnel syndrome
Synovium from surgery
Dairy farmer
2 months
Synovectomy
Full recovery
42 (M)
Synovitis
Synovectomy
Local steroid injection
12 months
14
47 (M)
Synovectomy
Nil
24 months
15
57 (F)
Carpal tunnel syndrome Ganglion
Synovial culture
Nil
24 months
16
63 (M)
Synovial culture
Nil
3 months
17
48 (F)
Flexor synovitis and carpal tunnel syndrome Synovitis
Fluid culture
Nil
3 months
18
52 (M)
TB 20 years previously Nodule biopsy from Skin laceration finger Synovial culture Skin laceration; steroid injection ?SLE Aspiration
13
[12]
19
[10]
55 (M)
Carpal tunnel syndrome Flexor tendinitis
20
[2]
31 (M)
Tendinitis
48 (F) 16 (M)
Carpal tunnel syndrome; synovitis Synovium Swelling of palm
23
44 (M)
Synovitis
Synovium
24
52 (M)
Synovitis
25
63 (F)
26 27
21 22
[3]
* Kettering.
Minor residual disability Limited range of movement. No pain Full recovery
and
Fluid aspiration
24 months 3 months 15 months Not known 4 months
Synovium
Oral steroids; lymphoma Non-penetrating trauma Oral steroids
Synovitis
Synovial culture
Nil
12 months
43 (M)
Tenosynovitis
Synovial culture
48 months
72 (M)
Carpal tunnel syndrome
Synovium
Non-penetrating trauma Penetrating trauma
24 months 2 months
24 months
chemotherapy Surgery; rifampicin; erythromycin Surgery and chemotherapy Chemotherapy alone Chemotherapy alone Surgery and chemotherapy Chemotherapy alone Chemotherapy alone Surgery and chemotherapy Surgery and chemotherapy Surgery and chemotherapy Surgery and chemotherapy Surgery and chemotherapy Surgery and chpmothcrflnv Surgery and chemotherapy Surgery and chemotherapy
Complete remission Complete remission Full remission Full remission Full remission Synovial thickening Full recovery Full recovery Full recovery Good recovery Slight stiffness Good recovery Good recovery Good recovery Not known
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5
1 month Advanced degenerative arthritis and steroid injection 12 months Steroid injection
DILLON ETAL.: M. KANSASII IN WRIST AND HAND
REFERENCES
1. Kelly PJ, Karlson AG, Weed LA, et al. Infection of
synovial tissues by mycobacteria other than mycobacterium tuberculosis. J Bone Joint Surg [Am] 1967; 49: 1521-30. 2. GuntherSF, Elliot RC. Mycobacteriumi kansasii infection in the deep structures of the hand. J Bone Joint Surg [Am] 1976; 58: 140-2. 3. Sutker WL, Lankford LL, Tompsett R. Granulomatous synovitis: the role of atypical mycobacterium. Rev Infect Dis 1979; 1: 729-35. 4. Dorff G, Frerichs L, Zabransky R, Spankus J. Musculo-skeletal infection due to Mycobacterium kansasii. Clin Orthop 1978; 136: 244-6. 5. Saphyakhajon P, Mukhopadhyay D, Spiegel P. Mycobacterium kansasii arthritis of the knee joint. Am J Dis Child 1977; 131: 573-6. 6. Godwin MC. Infection of knee joint by Mycobacterium kansasii. JAMA 1965; 194: 88-9. 7. De Merieux P, Keystone EC, Hutcheon M, Laskin C. Polyarthritis due to Mycobacterium kansasii in a patient with rheumatoid arthritis. Ann Rheum Dis 1980; 39: 90-4. 8. Chapman JS, Bernard JS, Speight M. Isolation of mycobacteria from raw milk. Am Rev Resp Dis 1965; 91: 351-5. 9. Chapman JS, Dyerly M, Spohn S. Epidemiological notes on Mycobacterium kansasii disease. Arch Environ Health 1968; 16: 673-8. 10. 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: 557-9. 11. Kaplan H, Clayton M. Carpal tunnel syndrome secondary to Mycobacterium kansasii infection. JAMA 1969; 208: 1186-8. 12. Dixon JH. Non-tuberculous mycobacterial infection of the tendon sheaths in the hand. J Bone Joint Surg [Br] 1981; 63: 542-4. 13. Gunther SF, Elliot RC, Brand RL, et al. Experience with atypical mycobacterial infection in the deep structures of the hand. / Hand Surg 1977; 2: 90-6. 14. Leader M, Revell P, Clarke G. Synovial infection with Mycobacterium kansasii. Ann Rheum Dis 1984; 43: 80-2. 15. Klinenberg JR, Grimley PM, Seegmiller JE. Destructive polyarthritis due to photochromogenic mycobacterium. N Engl J Med 1965; 272: 190-3. 16. Personal communication from the TB Reference Laboratory, Cardiff.
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There is considerable variation in the management of these patients; some authors have advocated surgery alone [1], whilst others have successfully used chemotherapy alone [10, 11]. Mycobacterium kansasii are notoriously resistant and some authors advise starting chemotherapy only when full sensitivities are known [14]. All of our patients received a combination of surgical excision with appropriate chemotherapy once sensitivities were known. The adequate length of treatment is not known but we have used 18 months [14]. The current recommendations for atypical pulmonary Mycobacterium used in the recent British Thoracic Society trial are ethambutol and rifampicin for 9 months. It is interesting to note that several authors have had a good clinical effect with erythromycin [4, 15], although results are not consistent. Why does there seem to be a pocket of synovial Mycobacterium kansasii infection in a small area of the Midlands? Is there, perhaps, a higher rate of atypical pulmonary mycobacterial infection locally? There is no evidence to support this [16], but perhaps this type of soft tissue infection is more common than previously thought and perhaps it is not being recognized. Certainly, in two of our cases the diagnosis was not considered early on and specimens were not sent for mycobacterial culture. Locally, specimens are sent for histology at the discretion of the surgeon, but the number arriving at the microbiology laboratory is very small. The experience of these cases suggests that many more, if not all, biopsy specimens of wrist and hand tenosynovitis and joint synovitis should go for culture on Lowenstein-Jensen or equivalent media, particularly if a granulomatous process is found at histology. In conclusion, Mycobacterium kansasii should be considered in a patient with a grumbling joint or tendon inflammation, an immunosuppressed patient or a patient with an established arthropathy that quickly worsens. Joint aspiration and synovial biopsy are indicated early, with specimens being sent for mycobacterial culture.
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