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REVIEW ARTICLES
Infectious Causes of Carpal Tunnel Syndrome: Case Report and Review John R. Mascola and Leland S. Rickman*
From the Infectious Diseases Division, National Naval Medical Center, Bethesda, Maryland
Carpal tunnel syndrome (CTS) is the name commonly ascribed to entrapment neuropathy of the median nerve at the wrist [1]. Compression occurs in the carpal tunnel of the hand where the median nerve and flexor tendons pass through a common tunnel bound on the sides by carpal bones and superficially by the transverse carpal ligament. Any process that alters the structures in the carpal tunnel can compress the median nerve, leading to a characteristic group of symptoms and physical findings. Nerve fibers that extend to the muscles of the thumb and provide sensation to the thumb, index, middle, and one-half ofthe ring finger originate in the median nerve; thus its entrapment is characterized by pain or paresthesias in these fingers and weakness or clumsiness in the use of the thumb. Physical findings include loss of sensation along the distribution of the median nerve and atrophy of the thenar muscles. CTS is a relatively common disorder that affects rvO.1 % of the population [2]. A variety of disorders may give rise to this syndrome, including trauma, rheumatoid arthritis, gout, myxedema, acromegaly, pregnancy, amyloidosis, and infection [3-6]. Although attention has most recently focused on the relatively common entity of work-related CTS [7], infection represents an important and treatable cause of CTS. We have recently seen a case of recurrent CTS that was caused by infection with Histoplasma capsulatum. The patient did well after surgery and administration of antifungal therapy,
Received 18 June 1990; revised 16 October 1990. * Present affiliation: University of California, San Diego, Medical Center H-208, 225 Dickinson Street, San Diego, California 92103. Reprints and correspondence: Dr. John R. Mascola, Infectious Diseases Division, National Naval Medical Center, Bethesda, Maryland 20889. Reviews of Infectious Diseases 1991;13:911-7 This article is in the public domain.
a situation which prompted us to review the infectious causes of CTS.
Case Report A 28-year-old male naval flight mechanic was hospitalized in October 1989 following his third episode of left-sided CTS in 18 months. During the initial two episodes, he had been treated with rest and nonsteroidal anti-inflammatory agents; the symptoms resolved over 2-3 weeks. On the occasion of the third episode, the patient presented with swelling of the wrist and hand and pain and numbness in the radial three fingers. These symptoms became worse at night, often awakening him from sleep. Initial conservative therapy and injection of steroids several weeks later failed to relieve the symptoms. Physical examination was remarkable only for sensory loss along the distribution of the median nerve and mild swelling over the lateral, palmar aspect of the hand. Electromyographic and nerve conduction studies revealed significant compromise of the median nerve at the wrist. The results of routine laboratory tests and roentgenograms of the chest and wrist were within normal limits. The patient had grown up in Kentucky and was most recently stationed in western Pennsylvania. There was no history of trauma to the left wrist. In October 1989 the patient underwent surgical exploration of the wrist that revealed a grossly thickened synovium surrounding the flexor tendons in the carpal tunnel. Synovectomy and lengthening of the transverse carpal ligament were performed. Microscopic examination of the synovium revealed chronic proliferative synovitis with fibrinoid necrosis and the presence of giant cells. Acid-fast and Grocott-Gomori methenamine-silver nitrate stains of the tissue revealed no organisms; fungal culture of the tissue yielded H. capsulatum. Therapy with ketoconazole (400 mg daily) was begun and was continued for 6 months. The patient recovered unevent-
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A case of carpal tunnel syndrome caused by Histoplasma capsulatum is presented and the medical literature is reviewed for cases of carpal tunnel syndrome that resulted from infection. Over 100 cases of carpal tunnel syndrome secondary to fungal, bacterial, mycobacterial, viral, and parasitic infections are reviewed. In most of these cases, infectious causes of the syndrome were not suspected; the etiology was often determined incidentally at surgery. Although infection is not a common cause of carpal tunnel syndrome, it should be considered in patients with persistent or recurrent symptoms. For those patients who require surgical exploration or carpal tunnel release, specimens should be submitted for histopathologic evaluation and culture. Most patients respond well to appropriate chemotherapy or surgery plus chemotherapy.
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fully; at a follow-up visit at 6 months, he was observed to be asymptomatic with full function of the left hand. Methods
Results and Discussion Infection is a relatively uncommon cause of CTS. In reviews of 439 and 329 cases of CTS, respectively, Phalen [1] and Ariyan and Watson [8] did not mention infectious causes of this disorder. However, in Phalen's series only 40 % of patients underwent surgery, and in neither report were the results of histologic studies or culture specifically described. In three other series of cases of CTS in which 342 surgical specimens were examined microscopically, three patients were identified as having presumed tuberculous tenosynovitis and one was identified as having nonspecific granulomatous synovitis [6, 9, 10]. We do not know if cultures were also performed on the surgical specimens. As the methodologies of these published studies did not include procedures for the identification of infectious etiologies of CTS, the true prevalence of such causes is unknown. Infectious Causes of CTS
In our review, 122 cases of infection-related CTS were identified that were caused by 22 different species of organisms. Etiologic agents included fungi, bacteria, mycobacteria, viruses, and a parasite. H. capsulatum. Including the case in our report, there have been six cases of CTS due to H. capsulatum infection reported inthe literature (table 1). A marked similarity in clinical presentation exists among the patients described in that they all experienced chronic or recurrent symptoms over months to years before the diagnosis was made. All patients were from areas that are endemic for H. capsulatum and only one patient (4) was immunocompromised. Three of the six patients were initially treated with surgery alone and all three experienced recurrent symptoms that resolved after they received
therapy with amphotericin B. The remaining three patients underwent surgery, received antifungal therapy, and did well. Our case is the only one reported in which ketoconazole rather than amphotericin B was used. Coccidioides immitis. Several articles have mentioned C immitis as a cause of CTS [4, 5, 15, 16]. However, the two reports that specifically describe coccidioidal tenosynovitis of the hand do not include findings that are specific for CTS [17, 18]. No other reports of cases of CTS due to C immitis were found in the literature. Sporothrix schenckii. S. schenckii is a saprophytic dimorphic fungus that most often causes a chronic subcutaneous infection that spreads via the lymphatic system. Occasionally, deep structures such as tenosynovium and bone may be involved [19, 20]. The organism is widespread in nature, existing in soil and on living and decaying plants. Infection in humans is often due to trauma and therefore gardeners, farmers, foresters, and those with related occupations are at greatest risk [19-21]. Six cases of CTS secondary to S. schenckii infection are described in table 2. Four of the six patients had a history of occupational exposure. Another had a history of alcoholism, in itself a predisposing condition [25]. Only one of the six patients presented with the classic nodular subcutaneous lesions; the remainder experienced CTS alone or in association with swelling of the hand or wrist. Treatment was problematic in that all six patients experienced recurrent symptoms after initial surgery (three had also received potassium iodide). After the recurrence of symptoms, one patient was again treated with potassium iodide and the remaining five were treated with amphotericin B. At follow-up, all six patients were doing well. The poor response of articular sporotrichosis to surgical therapy alone has been noted previously [19, 20, 23]. Therefore, surgical therapy has often been combined with the administration of potassium iodide or amphotericin B. Although controlled trials have not been conducted, recent reviews on articular sporotrichosis have suggested that administration of amphotericin B alone may be therapeutically sufficient [26, 27]. Four of the patients whose cases are reviewed in this report were treated medically with amphotericin B after surgical therapy failed. All four did well, which suggests that amphotericin B may be used as primary therapy in these cases. Mycobacterium tuberculosis. M. tuberculosis is cited most frequently as an infectious cause of CTS; there are 26 references in the literature that describe the etiologic role of this organism in CTS. Several reports on series of cases of tuberculosis of the hand and wrist have made reference to CTS [28-31]. Mason [29] reviewed 23 cases of M. tuberculosis infection of the hand and commented that involvement of the median nerve was "frequent," while in their review of 37 cases, Bickel et al. [30] found CTS to be "not uncommon." Six cases of CTS are included in a report on a series of cases of tuber-
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We reviewed the medical literature (English- and foreignlanguage articles) for reports of cases of CTS caused by infection. A computerized search was performed with use of the National Library of Medicine's MED LINE database to identify recent cases, and earlier cases were identified from the references in these more recent reports. Reports of series of cases of CTS were reviewed as were reports of series of cases with an emphasis on infections of the hands or wrists. For the purposes of this review, cases of CTS were included if the reports documented subjective and objective findings that were consistent with compression of the median nerve at the wrist and if they documented the identification of an organism by microscopic examination or culture.
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Table 1. Characteristics of six patients with CTS due to H. capsulatum. Patient no. [reference]
Sex/age (y)
Duration of symptoms
Predisposing condition
Positive tests *
Primary treatment
Recurrence
Secondary treatment
1 [11] 2 [12] 3 [13] 4 [14] 5 [15] 6 [PR]
M/24 M/32 M/24 F/43 F/43 M/28
Several mo 1Y 7 mo 11 mo 1Y 1.5 Y
No No No Yest No No
Culture, SS Culture Culture, SS SS Culture, SS Culture
Surgery Surgery, AmB (3.5 g) Surgery Surgery, AmB (1 g) Surgery Surgery, Ket
Yes No Yes No Yes No
Surgery, AmB (1.6 g) None Surgery, ~mB None ArnB, (l g) None
NOTE. SS = Grocott-Gomori methenamine-silver nitrate stain; AmB = amphotericin B; PR * Results of culture and histopathologic examination of surgical tissue specimen. t Patient had a history of breast cancer and recent chemotherapy.
Outcome Well at 4 Well at 2 Did well Well at 4 Well at 4 Well at 6
y y mo mo rno
= present report; Ket = ketoconazole.
Patient no. [reference]
Sex/age (y)
1 [22] 2 [22] 3 [23] 4 [24] 5 [25] 6 [25]
F/58 M/65 M/54 M/49 M/43
M/49
Risk factor
Positive culture
Primary treatment
Recurrence
Secondary treatment
Gardening Farmer None Tree cutter Farmer Alcoholism
Yes Yes Yes Yes Yes Yes
Surgery, KI Surgery Surgery, KI Surgery, KI Surgery Surgery
Yes Yes Yes Yes Yes Yes
Surgery, KI AmB (943 mg) AmB (450 mg) AmB (3.8 g) AmB (1 g) Surgery, AmB (1.5 g)
Outcome Well Well Well Well Well Well
at at at at at at
10 y 7 mo 6w 2y 4y 4y
NOTE. KI = potassium iodide; AmB = amphotericin B.
Table 3. Characteristics of nine patients with tuberculous CTS. Patient no. [reference]
Sex/ age (y)
Positive skin test
1 [38]
M/57
Yes
2 [39]
M/44
Yes
3 [40] 4 [40]
M/66
M/66
ND ND
5 [41]
F/46
Yes
Miliary tuberculosis, + gastric aspirate Hilar adenopathy, axillary node + for acid-fast bacilli Tuberculosis in 1940 Tuberculosis of the spine, age 18 No
6 [5]
M/50
Yes
No
7 [5]
M/66
Yes
Old perihilar scarring
8 [42] 9 [43]
M/41 F/72
No No
No No
Evidence of systemic disease
Other positive tests *
Primary treatment
AFS Culture Culture
Surgery, INH, PAS, STM Surgery, INH, RMP
No
None
Did well
No
None
Well at 7 w
Culture Culture
Surgery, INH, RMP Surgery, INH, EMB
No No
None None
Well at 1 y Well at 6 mo
Culture
Surgery, INH, RMP, EMB Surgery, INH, RMP, EMB Surgery, INH, EMB
No
None
Well at 12 mo
No
None
Fusion of wrist
No
None
Surgery Surgery
Yes Yes
Surgery, INH, RMP Surgery, chemotherapy
Flexion deformity of wrist Did well Did well
AFS Culture AFS Culture Culture Culture
NOTE. AFS = acid-fast stain; INH = isoniazid; PAS = p-aminosalicylic acid; STM * Refers to culture and histopathologic examination of surgical tissue specimen.
culous arthritis [32], and eight other cases of tuberculous arthritis are mentioned in a report on a large series of cases ofCTS [6,9, 10,33,34]. Specific details regarding the cases are not given in these reports. Three reports from the foreignlanguage literature also mention CTS due to infection with
Recurrence Secondary treatment
Outcome
= streptomycin; RMP = rifampin; ND = note done; EMB = ethambutol.
M. tuberculosis [35-37]. The remaining nine cases are summarized in table 3. It is believed that peripheral tuberculous arthritis results from reactivation of a latent focus of infection rather than from direct inoculation of the organisms [32]. Moreover, eight of
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Table 2. Characteristics of six patients with CTS due to S. schenckii.
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responded to treatment with isoniazid, ethambutol, and rifampin. Treatment in the majority of these cases consisted of surgery and the administration of chemotherapy. Patients generally did quite well and experienced complete resolution of symptoms. Only three of 22 patients were experiencing mild residual functional deficits at follow-up. Since chemotherapeutic combinations varied according to the infecting species of Mycobacterium, the reader is referred to the original references for details regarding specific treatment regimens. Bacterial causes ofCTS. There have been eight reported cases of acute pyogenic hand infections that resulted in CTS (table 4). Etiologic organisms include Staphylococcus aureus, coagulase-negative staphylococci, t1-hemolytic streptococci, Enterococcus faecalis, and Clostridium histolyticum. In marked contrast to patients with CTS caused by fungal or mycobacterial infection, all eight of these patients experienced some residual functional or sensory deficit after treatment. Not included in table 4 are three cases of CTS secondary to infection with Neisseria gonorrhoeae [65-67]. One patient had a clinical syndrome consistent with disseminated gonorrhea; however, on presentation, two others were asymptomatic except for the symptoms of CTS. Cultures of surgical specimens in both cases yielded N. gonorrhoeae. Disseminated gonococcal infection that manifests solely as monoarticular disease has been reported previously [68]. Borrelia burgdorferi. Infection with B. burgdorferi has recently been reported to cause CTS [68]. In a prospective study of patients with late-stage Lyme disease, Halperin et al. [69] found that 19 of 76 patients had symptoms of intermittent hand paresthesias, including 14 who had neurophysiologic evidence of entrapment of the median nerve at the wrist. All patients had been referred to these authors for evaluation of systemic or rheumatologic problems; in none of these cases were the neurologic symptoms the patient's primary problem. Clinical evidence of hand tenosynovitis or arthritis was present in two of the patients. Thus, mild cases of CTS may be fairly common in patients who have late-stage Lyme disease. Finally, toxic shock syndrome has also been associated with
Table 4. Characteristics of eight patients with bacterial CTS. Patient no. [reference]
1 [60] 2 [60] 3 [61] 4 [61] 5 [61] 6 [62] 7 [63] 8 [64]
Sexl age (y)
Trauma to hand
Culture results
Treatment
Outcome
F/58 F/53 F/68 M/52 F/50 F/IO M/23 M/24
Yes Yes No Yes Yes Yes No Yes
CN-Staph Sterile pus p-strep Polymicrobial * Sterile exudate Staphylococcus aureus Clostridium histolyticum S. aureus
Surgery, antibiotics Surgery, antibiotics Surgery, antibiotics Surgery, antibiotics .Surgery, antibiotics Surgery, antibiotics Surgery, antibiotics Aspiration, antibiotics
Mild motor and sensory deficits Persistent paresthesias Mild sensory deficit Mild functional and sensory deficits Slight sensory deficit Mild sensory deficit Mild functional impairment Significant median nerve palsy
NOTE. eN-Staph = coagulase-negative staphylococci; l3-strep = l3-hemolytic streptococci. * S. aureus; Enterococcus faecalis; l3-hemolytic streptococci.
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nine patients who are described in table 3 had a history of a reactive tuberculin test or an abnormal chest roentgenogram. Evidence of active disease was noted for two of these patients. Treatment of peripheral tuberculous arthritis has generally consisted of surgery and the administration of antituberculous medications, with good results [32,40]. In this review, seven of nine patients who were treated surgically and received chemotherapy did well. Two patients underwent surgery and did not receive medical treatment; both experienced recurrent episodes of CTS. Other Mycobacterium species. In reviewing the literature, we found 23 reported cases of CTS due to nontuberculous mycobacteria. The species implicated in these cases included Mycobacterium marinum (7 cases) [44-46]; the Mycobacterium avium complex (5 cases) [47-49]; Mycobacterium kansasii (4 cases) [47, 50-52]; Mycobacterium chelonei [53]; Mycobacterium fortuitum [14]; Mycobacterium szulgai [16]; Mycobacterium terrae [54]; Mycobacterium malmoense [55]; Mycobacterium leprae [56]; and Mycobacterium bovis [57]. Most cases occurred in patients with a history of exposure to the organism (e.g., contaminated water or fish infected with M. marinum) or a history of minor trauma to the hand. Only two patients had an underlying immunocompromising condition. Several of these cases deserve comment. The patient with CTS secondary to infection with M. leprae had no cutaneous manifestations of leprosy. In this case the diagnosis was made by visualization of acid-fast organisms that were isolated from a biopsy specimen of the median nerve, on the basis of a positive lepromin test, and on the basis of clinical response to dapsone. M. leprae is the only mycobacterium that specifically affects the peripheral nerves, and involvement of the median nerve is a common manifestation of leprosy [56]. However, this rarely occurs in the absence of ulnar neuropathy [56-59], and therefore this case of CTS represents a rare manifestation of leprosy. The M. bovis infection occurred in a patient who stuck herselfwith a Pasteur pipette that contained the Calmette-Guerin bacillus. CTS developed several weeks later and the patient
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Summary We have reviewed the known infectious causes of CTS, including bacteria, mycobacteria, fungi, viruses, and parasites. These are summarized in table 5. In most of the cases described, an infectious etiology was not suspected until it became apparent incidentally on surgical exploration or carpal tunnel release. The pathogenesis of CTS was not clear in all cases. Some organisms directly infect the tissues of the carpal tunnel, multiply, and compress the median nerve. Infection may occur via hematogenous spread (N. gonorrhoeae) or direct inoculation (sporotrichosis, M. marinum). In systemic illnesses such as rubella and toxic shock syndrome, it is possible that CTS results from an immune response to in-
Table 5.
Summary of infectious causes of CTS.
Cause of CTS
Histoplasma capsulatum Sporothrix schenckii Mycobacterium tuberculosis MOTT Pyogenic bacteria* Neisseria gonorrhoeae Borrelia burgdorferi TSS Rubella virus Rubella vaccine Guinea worm
No. of cases
Reference
6 6 26 23 8 4 14 4
[11-15] [22-25] [6, 9, to, 32-43] [16, 44-57] [60-64] [65-67] [69] [70] [77-82] [83-86] [89]
13 17 1
NOTE. MOTT = mycobacteria other than tuberculosis; TSS syndrome. * See text for specific organisms.
=
toxic shock
fection or the production of an exotoxin. Bilateral CTS is more common in patients with these systemic illnesses. Although the exact prevalenceof infectious CTS is unknown, infection should be considered in cases of prolonged or recurrent CTS. If symptoms are severe enough to warrant carpal tunnel release, tissue specimens should be obtained for microscopic evaluation and culture. Most patients with infectious CTS respond well to appropriate chemotherapy or surgery plus chemotherapy. Acknowledgment The authors thank Judy Emmons for assistance in the preparation of this manuscript.
References 1. Phalen GS. The carpal-tunnel syndrome. Seventeen years' experience in diagnosis and treatment of six hundred fifty-four hands. J Bone Joint Surg [Am] 1966;48A:211-28 2. Katz JN, Larson MG, Sabra A, Krarup C, Stirrat CR, Sethi R, Eaton HM, Fossel AH, Liang MH. The carpal tunnel syndrome: diagnostic utility of the history and physical examination findings. Ann Intern Med 1990;112:321-7 3. Nakano KK. Entrapment neuropathies. In: Kelley WN, Harris ED Jr, Ruddy S, Sledge CB, eds. Textbookof rheumatology, 3rd ed. Philadelphia: WB Saunders, 1989:1845--59 4. Biundo n. Regional rheumatic pain syndromes. In: Primer on the rheumatic diseases. Atlanta: Arthritis Foundation, 1988:263-74 5. Klofkorn RW, Steigerwald Je. Carpal tunnel syndrome as the initial manifestation of tuberculosis. Am J Moo 1976;60:583-6 6. Inglis AE, Straub LR, Williams CS. Median nerve neuropathy at the wrist. Clin Orthop 1972;83:48-54 7. Baker EL, Ehrenberg RL. Preventing the work-related carpal tunnel syndrome: physician reporting and diagnostic criteria [editorial]. Ann Intern Med 1990;112:317-9 8. Ariyan S, Watson HK. The palmar approach for the visualization and release of the carpal tunnel. An analysis of 429 cases. Plast Reconstr Surg 1977;60:539-47
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cases of CTS. Sahs et al. [70] reported four patients with toxic shock syndrome who experienced bilateral CTS during their illness. Symptoms resolved slowly in all patients. Although neuromuscular sequelae have been described in patients with toxic shock syndrome [71], the pathogenesis of CTS in patients with this disorder is not known. Rubella-induced CTS. Arthritis is a relatively common complication of rubella that occurs most often in young women. The hands, wrists, and knees are affected most frequently [72]. In 1940 several letters appeared in the British Medical Journal that described pain, numbness, and weakness of the hands of women with rubella [73-76]. Although these cases were not described in enough detail to document the presence of CTS, there have been 13 subsequent reports of cases of CTS associated with rubella [77-82] and 17 cases have occurred following immunization against rubella [83-86] (13 adults and 17 children). All cases that occurred in adults were in young women and the symptoms almost always occurred bilaterally. In children the symptoms were usually bilateral as well, but in contrast to adults, both sexes were affected. In only one instance did a patient undergo surgery for carpal tunnel release. Symptoms resolved over several weeks in all cases. Hepatitis B virus. Massey et a1. [87] described a 29-yearold woman who had a protracted hepatitis B virus infection and right-sided CTS. However, CTS did not develop until the sixth month of illness and there were no other signs of arthritis. Therefore, in this case we cannot be sure that CTS was related to the hepatitis infection. Guinea worm infestation. The guinea worm, or Dracunculus medinensis, is acquired by drinking water that contains crustaceans which have ingested the larvae of this nematode [88]. A report from Madras, India, described a 55-year-old woman with guinea worm infestation of the right wrist that caused CTS [89]. At surgical exploration, a calcified guinea worm was observed among the flexor tendons, adhering to the median nerve. After the parasite was removed the patient did well.
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37. Mark G, Gartmann J, Ruedi T. "Karpaltunnelsyndrorne" bei Seh nenscheiden tuberkulose der Hand. Schweiz Med Wochenschr 1984; 114:448-51 38. Mayers LB. Carpal tunnel syndrome secondary to tuberculosis. Arch Neurol 1964;10:426-9 39. Lee KE. Thbcrculosis presenting as carpal tunnel syndrome. J Hand Surg [Am] 1985;10A:242-5 40. Bush DC, Schneider LH. Tuberculosis of the hand and wrist. J Hand Surg [Am] 1984;9A:391-8 41. Gouet P, Castets M, Touchard G, Payen J, Alcalay M. Bilateral carpal tunnel syndrome due to tuberculosis tenosynovitis: a case report [letter]. J Rheumatol 1984;11:721-2 42. Suso S, Peidro L, Ramon R. Tuberculous synovitis with "rice bodies" presenting as carpal tunnel syndrome. J Hand Surg [Am) 1988;13A: 574-6 43. Langa V, Posner MA, Hoffman S, Steiner Ge. Carpal tunnel syndrome secondary to tuberculous tenosynovitis. Bull Hosp Jt Dis Orthop Inst 1986;46:137-42 44. Hurst LC, Amadio PC, Badalamente MA, Ellstein JL, Dattwyler RJ. Mycobacterium marinum infections of the hand. J Hand Surg [Am] 1987;12A:428-35 45. Williams CS, Riordan De. Mycobacterium marinum (atypical acid-fast bacillus) infections of the hand: a report of six cases. ] Bone Joint Surg [Am] 1973;55A:1042-50 46. Chow SP, Ip FK, Lau JH, Collins RJ, Luk KD, So YC, Pun WK. Mycobacterium marinum infections of the hand and wrist. Results of conservative treatment in twenty-four cases. J Bone Joint Surg 1987;69A:1161-8 47. Kelly PJ, Karlson AG, Weed LA, Lipscomb PRo Infection of synovial tissues by mycobacteriaother than Mycobacterium tuberculosis. J Bone Joint Surg [Am] 1967;49A:1521-30 48. Cheatum DE, Hudman V, Jones SR. Chronic arthritis due to Mycobacterium intracellulare. Sacroiliac, knee, and carpal tunnel involvement in a young man and response to chemotherapy. Arthritis Rheum 1976;19:777-81 49. Sutker WL, Lankford LL, Tompsett R. Granulomatous synovitis: the role of atypical mycobacteria. Rev Infect Dis 1979;1:729-35 50. Kaplan H, Clayton M. Carpal tunnel syndrome secondary to Mycobacterium kansasii infection. JAMA 1969;208:1186-8 51. Gunther SF, Elliott Re. Mycobacterium kansasii infection in the deep structures of the hand. Report of two cases. J Bone Joint Surg [Am] 1976;58A:140-2 52. Dorff GJ, Frerichs L, Zabransky RJ, Jacobs P, SpankusJD. Musculoskeletal infections due to Mycobacterium kansasii. Clinical Orthopaedics and Related Research 1978;136:244-6 53. Zachary LS, Clark GL Jr, Kleinert JM, O'Donovan C III. Mycobacterium chelonei tenosynovitis. Ann Plast Surg 1988;20:360-2 54. Deenstra W. Synovial hand infection from Mycobacterium terrae. J Hand Surg [Br] 1988;13B:335-6 55. Prince H, Ispahani P, Baker M. A Mycobacterium malmoense infection of the hand presenting as carpal tunnel syndrome. J Hand Surg [Br] 1988;13B:328-30 56. Selby Re. Neurosurgical aspects of leprosy. Surg NeuroI1974;2:165-77 57. Janier M, Gheorghiu M, Cohen P, Mazas F, Duroux P. Synrome du canal carpien a mycobacterium bovis BCG. Sem Hop Paris 1982; 58:977-9 58. Calloway JM, Fite GL, Riordan De. Ulnar and median neuritis due to leprosy. Int J Lepr Other Mycobact Dis 1964;32:285-91 59. Brand PW. Treatment of Leprosy. II. The role of surgery. N Engl J Med 1956;254:64-7 60. Bailey D, Bolton Carter JE Median-nerve palsy associated with acute infections of the hand. Lancet 1955;268:530-2 61. Oates GD. Median-nerve palsy as a complication of acute pyogenic in-
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9. Yamaguchi DM, Lipscomb PR, Soule EH. Carpal tunnel syndrome. Minn Med 1965;48:22-33 10. Mangini U. Some remarks on the etiology of the carpal tunnel compression of the median nerve. Bull Hosp Jt Dis Orthop Inst 1961;22:56-64 11. Perlman R, Jubelirer RA, Schwarz J. Histoplasmosis of the common palmar tendon sheath. J Bone Joint Surg [Am] 1972;54A:676-8 12. Orner GE Jr, Lockwood RS, Travis LO. Histoplasmosis involving the carpal joint: a case report. J Bone Joint Surg [Am] 1963;45A: 1699-1703 13. Vanek J, Schwarz J. The gamut of histoplasmosis. Am J Med 1971;50: 89-104 14. Randall G, Smith PW, Korbitz B, Owen DR. Carpal tunnel syndrome caused by Mycobacterium fortuitum and Histoplasma capsulatum. Report of two cases. J Neurosurg 1982;56:299-301 15. Strayer OS, Gutwein MB, Herbold D, Bresalier R. Histoplasmosis presenting as the carpal tunnel syndrome. Am J Surg 1981;141:286-8 16. Stratton CW, Phelps DB, Reller LB. Thberculoid tenosynovitis and carpal tunnel syndrome caused by Mycobacterium szulgai. Am J Med 1978;65:349-51 17. Iverson RE, Vistnes LM. Coccidioidomycosis tenosynovitis in the hand. J Bone Joint Surg [Am] 1973;55A:413-7 18. Walker OR, Hall RH. Coccidioidal tenosynovitis: report of a case. J Bone Joint Surg [Am] 1954;36A:391-2 19. WIlsonDE, Mann n, Bennett JE, Utz JP. Clinical featuresof extracutaneous sporotrichosis. Medicine (Baltimore) 1967;46:265-79 20. Bayer AS, Scott VJ, Guze LB. Fungal arthritis. III. Sporotrichal arthritis. Semin Arthritis Rheum 1979;9:66-74 21. Bennett JE. Sporothrix Schenckii. In: Mandell GL, Douglas RG, Bennett JE, eds. Principles and practice of infectious diseases. New York: Churchill Livingstone, 1990:1972-5 22. Hagemann PO. Sporotrichosis tendonitis and tenosynovitis. Trans Am Clin Climatol Assoc 1967;79:193-8 23. Altner PC, Thrner RR. Sporotrichosis of bones and joints: review of the literature and report of six cases. Clin Orthop 1970;68:138-48 24. Weitzner R, Mak E, Lertratanakul Y. Articular sporotrichosis [letter]. Ann Intern Med 1977;87:382 25. Stratton CW, Lichtenstein KA, Lowenstein SR, Phelps DB, Reller LB. Granulomatous tenosynovitis and carpal tunnel syndrome caused by Sporothrix schenckii. Am J Med 1981;71:161-4 26. Crout JE, Brewer NS, Tompkins RB. Sporotrichosis arthritis: clinical features in seven patients. Ann Intern Med 1977;86:294-7 27. Winn RE. Sporotrichosis. Infect Dis Clin North Am 1988;2:899-911 28. Kanavel AB. Thberculous tenosynovitis of the hand: a report of fourteen cases of tuberculous tenosynovitis. Surg Gynecol Obstet 1923;37: 635-47 29. Mason ML. Thberculous tenosynovitis of the hand: a study of thirtythree cases of chronic tenosynovitis of the hand. Surg Gynecol Obstet 1934;59:363-9 30. Bickel WH, Kimbrough RF, Dahlin De. Thbcrculous tenosynovitis. JAMA 1953;151:31-5 31. Adams R, Jones G, Marble He. Thbcrculous tenosynovitis. N Engl J Med 1940;223 :706-8 32. Garrido G, Gomez-Reino n, Fernandez-Depica P, Palenque E, Prieto S. A reviewof peripheral tuberculous arthritis. Semin Arthritis Rheum 1988;18:142-9 33. Vainio K. Carpal canal syndrome caused by tenosynovitis. Acta Rheumatol Scand 1958;4:22-7 .. 34. Scarff JE, Vicale CT. Compression of the median nerve in the carpal tunnel. Trans Am Neurol Assoc 1955;80:114-7 35. Ficat P, Arlet 1. Carpal tunnel syndrome due to tenosynovitis during chronic evolutive arthritis. Rev Rhum Mal Osteoartic 1956;23:17 36. Arlet J, Ficat P, Martinez 1. Carpal tunnel syndrome caused by tophaceous gout. Rev Rhum Mal Osteoartic 1958;25:581
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74. 75. 76. 77. 78. 79. 80.
81. 82. 83. 84. 85. 86.
87. 88. 89.
Harrison BL. Neuritis following rubella [letter]. B M I 1940;1:637 Simpson REH. Rubella and polyarthritis [letter). B M J 1940;1:830 Hodges GMW. Neuritis following rubella [letter]. B M J 1940;3:830-1 Fry I, Dillane JB, Fry L. Rubella, 1962. B M I 1962;3:833-4 Heathfield KWG. Carpal-tunnel syndrome [letter). B M J 1962;2:58 Ellis W. Rubella arthritis [letter]. B M J 1973;4:549 Blennow G, Bekassy AN, Eriksson M, Rosendahl R. Transient carpal tunnel syndrome accompanying rubella infection. Acta Paediatr Scand 1982;71:1025-8 Chambers RI, Bywaters EGL. Rubella synovitis. Ann Rheum Dis 1963;22 :263-8 Yanez IE, Thompson GR, Mikkelsen WM, Bartholomew LE. Rubella arthritis. Ann Intern Med 1966;64:772-7 Hale MS, Ruderman IE. Carpal tunnel syndrome associated with rubella immunization. Am J Phys Moo 1973;52:189-94 Cooper LZ, Ziring PR, Weiss HJ, Matters MA, Krugman S. Transient arthritis after rubella vaccination. Am J Dis Child 1969;118:218-25 Thompson GR, Ferreyra A, Brackett RG. Acute arthritis complicating rubella vaccination. Arthritis Rheum 1971;14:19-26 Tingle AI, Chantler JK, Pot KH, Paty OW, Ford OK. Postpartum rubella immunization: association with development of prolonged arthritis, neurological sequelae, and chronic rubella viremia. J Infect Dis 1985;152:606-12 Massey EW, Folger WN, Holohan T, Pellock KJ. Carpal tunnel syndrome in hepatic disease [letter). South Med I 1979;72:1030 Hopkins DR. Dracunculiasis. In: Warren KS, Mahmoud AF, eds. Tropical and geographic medicine. New York: McGraw-Hill, 1990:439-42 Balasubramanian V, Ramamurthi B. An unusual location of guineawonn infestation: report of a case. J Neurosurg 1965;23:537-8
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fections of the hand. B M I 1960;2:1618-20 62. Willians LF Ir, Geer T. Acute carpal tunnel syndrome secondary to pyogenic infection of the forearm. lAMA 1963;185:409-10 63. Gerardi lA, Mack GR, Lutz RB. Acute carpal tunnel syndrome secondary to septic arthritis of the wrist. I Am Osteopath Assoc 1989;89: 933-4 64. Lindsay SL, Kerridge R, Collett B-1. Abscess following cannulation of the radial artery. Anaesthesia 1987;42:654-7 65. DeHertogh 0, Ritland 0, Green R. Carpal tunnel syndrome due to gonococcal tenosynovitis. Orthopedics 1988;11:199-200 66. Balcomb TV, Acute gonococcal flexor tenosynovitis in a woman with asymptomatic gonorrhea-case report and literature review. I Hand Surg [Am] 1982;7:521-2 67. Barrick EE Acutegonococcal flexor tenosynovitisin a woman with asymptomatic gonorrhea [letter]. I Hand Surg [Am] 1983;8:224-5 68. Keiser H, Ruben FL, Wolinsky E, Kushner I. Clinical forms of gonococcal arthritis. N Engl I Med 1968;279:234-40 69. Halperin II, Volkman 01, Luft BI, Dattwyler RI. Carpal tunnel syndrome in Lyme borreliosis. Muscle Nerve 1989;12:397-400 70. Sahs AL, Helms CM, Dubois C. Carpal tunnel syndrome: complication of toxic shock syndrome. Arch Neurol 1983;40:414-5. 71. Chesney PI, Crass BA, Polyak MB, Wand PI, Warner TF, Vergeront 1M, Davis IP, Toffe RW, Chesney RW, Bergdoll MS. Toxic shock syndrome: management and long-term sequelae. Ann Intern Med 1982; 96:847-51 72. Schnitzer T1. Viral arthritis. In: Kelley WN, Harris ED Jr, Ruddy S, Sledge CB, eds. Textbook of rheumatology, 3rd ed. Philadelphia: WB Saunders, 1989:1611-28 73. Hodges GMW. Brachial neuritis following rubella [letter]. B M I 1940;1:548
917
REVIEW ARTICLES
Infectious Causes of Carpal Tunnel Syndrome: Case Report and Review John R. Mascola and Leland S. Rickman*
From the Infectious Diseases Division, National Naval Medical Center, Bethesda, Maryland
Carpal tunnel syndrome (CTS) is the name commonly ascribed to entrapment neuropathy of the median nerve at the wrist [1]. Compression occurs in the carpal tunnel of the hand where the median nerve and flexor tendons pass through a common tunnel bound on the sides by carpal bones and superficially by the transverse carpal ligament. Any process that alters the structures in the carpal tunnel can compress the median nerve, leading to a characteristic group of symptoms and physical findings. Nerve fibers that extend to the muscles of the thumb and provide sensation to the thumb, index, middle, and one-half ofthe ring finger originate in the median nerve; thus its entrapment is characterized by pain or paresthesias in these fingers and weakness or clumsiness in the use of the thumb. Physical findings include loss of sensation along the distribution of the median nerve and atrophy of the thenar muscles. CTS is a relatively common disorder that affects rvO.1 % of the population [2]. A variety of disorders may give rise to this syndrome, including trauma, rheumatoid arthritis, gout, myxedema, acromegaly, pregnancy, amyloidosis, and infection [3-6]. Although attention has most recently focused on the relatively common entity of work-related CTS [7], infection represents an important and treatable cause of CTS. We have recently seen a case of recurrent CTS that was caused by infection with Histoplasma capsulatum. The patient did well after surgery and administration of antifungal therapy,
Received 18 June 1990; revised 16 October 1990. * Present affiliation: University of California, San Diego, Medical Center H-208, 225 Dickinson Street, San Diego, California 92103. Reprints and correspondence: Dr. John R. Mascola, Infectious Diseases Division, National Naval Medical Center, Bethesda, Maryland 20889. Reviews of Infectious Diseases 1991;13:911-7 This article is in the public domain.
a situation which prompted us to review the infectious causes of CTS.
Case Report A 28-year-old male naval flight mechanic was hospitalized in October 1989 following his third episode of left-sided CTS in 18 months. During the initial two episodes, he had been treated with rest and nonsteroidal anti-inflammatory agents; the symptoms resolved over 2-3 weeks. On the occasion of the third episode, the patient presented with swelling of the wrist and hand and pain and numbness in the radial three fingers. These symptoms became worse at night, often awakening him from sleep. Initial conservative therapy and injection of steroids several weeks later failed to relieve the symptoms. Physical examination was remarkable only for sensory loss along the distribution of the median nerve and mild swelling over the lateral, palmar aspect of the hand. Electromyographic and nerve conduction studies revealed significant compromise of the median nerve at the wrist. The results of routine laboratory tests and roentgenograms of the chest and wrist were within normal limits. The patient had grown up in Kentucky and was most recently stationed in western Pennsylvania. There was no history of trauma to the left wrist. In October 1989 the patient underwent surgical exploration of the wrist that revealed a grossly thickened synovium surrounding the flexor tendons in the carpal tunnel. Synovectomy and lengthening of the transverse carpal ligament were performed. Microscopic examination of the synovium revealed chronic proliferative synovitis with fibrinoid necrosis and the presence of giant cells. Acid-fast and Grocott-Gomori methenamine-silver nitrate stains of the tissue revealed no organisms; fungal culture of the tissue yielded H. capsulatum. Therapy with ketoconazole (400 mg daily) was begun and was continued for 6 months. The patient recovered unevent-
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A case of carpal tunnel syndrome caused by Histoplasma capsulatum is presented and the medical literature is reviewed for cases of carpal tunnel syndrome that resulted from infection. Over 100 cases of carpal tunnel syndrome secondary to fungal, bacterial, mycobacterial, viral, and parasitic infections are reviewed. In most of these cases, infectious causes of the syndrome were not suspected; the etiology was often determined incidentally at surgery. Although infection is not a common cause of carpal tunnel syndrome, it should be considered in patients with persistent or recurrent symptoms. For those patients who require surgical exploration or carpal tunnel release, specimens should be submitted for histopathologic evaluation and culture. Most patients respond well to appropriate chemotherapy or surgery plus chemotherapy.
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Mascola and Rickman
fully; at a follow-up visit at 6 months, he was observed to be asymptomatic with full function of the left hand. Methods
Results and Discussion Infection is a relatively uncommon cause of CTS. In reviews of 439 and 329 cases of CTS, respectively, Phalen [1] and Ariyan and Watson [8] did not mention infectious causes of this disorder. However, in Phalen's series only 40 % of patients underwent surgery, and in neither report were the results of histologic studies or culture specifically described. In three other series of cases of CTS in which 342 surgical specimens were examined microscopically, three patients were identified as having presumed tuberculous tenosynovitis and one was identified as having nonspecific granulomatous synovitis [6, 9, 10]. We do not know if cultures were also performed on the surgical specimens. As the methodologies of these published studies did not include procedures for the identification of infectious etiologies of CTS, the true prevalence of such causes is unknown. Infectious Causes of CTS
In our review, 122 cases of infection-related CTS were identified that were caused by 22 different species of organisms. Etiologic agents included fungi, bacteria, mycobacteria, viruses, and a parasite. H. capsulatum. Including the case in our report, there have been six cases of CTS due to H. capsulatum infection reported inthe literature (table 1). A marked similarity in clinical presentation exists among the patients described in that they all experienced chronic or recurrent symptoms over months to years before the diagnosis was made. All patients were from areas that are endemic for H. capsulatum and only one patient (4) was immunocompromised. Three of the six patients were initially treated with surgery alone and all three experienced recurrent symptoms that resolved after they received
therapy with amphotericin B. The remaining three patients underwent surgery, received antifungal therapy, and did well. Our case is the only one reported in which ketoconazole rather than amphotericin B was used. Coccidioides immitis. Several articles have mentioned C immitis as a cause of CTS [4, 5, 15, 16]. However, the two reports that specifically describe coccidioidal tenosynovitis of the hand do not include findings that are specific for CTS [17, 18]. No other reports of cases of CTS due to C immitis were found in the literature. Sporothrix schenckii. S. schenckii is a saprophytic dimorphic fungus that most often causes a chronic subcutaneous infection that spreads via the lymphatic system. Occasionally, deep structures such as tenosynovium and bone may be involved [19, 20]. The organism is widespread in nature, existing in soil and on living and decaying plants. Infection in humans is often due to trauma and therefore gardeners, farmers, foresters, and those with related occupations are at greatest risk [19-21]. Six cases of CTS secondary to S. schenckii infection are described in table 2. Four of the six patients had a history of occupational exposure. Another had a history of alcoholism, in itself a predisposing condition [25]. Only one of the six patients presented with the classic nodular subcutaneous lesions; the remainder experienced CTS alone or in association with swelling of the hand or wrist. Treatment was problematic in that all six patients experienced recurrent symptoms after initial surgery (three had also received potassium iodide). After the recurrence of symptoms, one patient was again treated with potassium iodide and the remaining five were treated with amphotericin B. At follow-up, all six patients were doing well. The poor response of articular sporotrichosis to surgical therapy alone has been noted previously [19, 20, 23]. Therefore, surgical therapy has often been combined with the administration of potassium iodide or amphotericin B. Although controlled trials have not been conducted, recent reviews on articular sporotrichosis have suggested that administration of amphotericin B alone may be therapeutically sufficient [26, 27]. Four of the patients whose cases are reviewed in this report were treated medically with amphotericin B after surgical therapy failed. All four did well, which suggests that amphotericin B may be used as primary therapy in these cases. Mycobacterium tuberculosis. M. tuberculosis is cited most frequently as an infectious cause of CTS; there are 26 references in the literature that describe the etiologic role of this organism in CTS. Several reports on series of cases of tuberculosis of the hand and wrist have made reference to CTS [28-31]. Mason [29] reviewed 23 cases of M. tuberculosis infection of the hand and commented that involvement of the median nerve was "frequent," while in their review of 37 cases, Bickel et al. [30] found CTS to be "not uncommon." Six cases of CTS are included in a report on a series of cases of tuber-
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We reviewed the medical literature (English- and foreignlanguage articles) for reports of cases of CTS caused by infection. A computerized search was performed with use of the National Library of Medicine's MED LINE database to identify recent cases, and earlier cases were identified from the references in these more recent reports. Reports of series of cases of CTS were reviewed as were reports of series of cases with an emphasis on infections of the hands or wrists. For the purposes of this review, cases of CTS were included if the reports documented subjective and objective findings that were consistent with compression of the median nerve at the wrist and if they documented the identification of an organism by microscopic examination or culture.
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Table 1. Characteristics of six patients with CTS due to H. capsulatum. Patient no. [reference]
Sex/age (y)
Duration of symptoms
Predisposing condition
Positive tests *
Primary treatment
Recurrence
Secondary treatment
1 [11] 2 [12] 3 [13] 4 [14] 5 [15] 6 [PR]
M/24 M/32 M/24 F/43 F/43 M/28
Several mo 1Y 7 mo 11 mo 1Y 1.5 Y
No No No Yest No No
Culture, SS Culture Culture, SS SS Culture, SS Culture
Surgery Surgery, AmB (3.5 g) Surgery Surgery, AmB (1 g) Surgery Surgery, Ket
Yes No Yes No Yes No
Surgery, AmB (1.6 g) None Surgery, ~mB None ArnB, (l g) None
NOTE. SS = Grocott-Gomori methenamine-silver nitrate stain; AmB = amphotericin B; PR * Results of culture and histopathologic examination of surgical tissue specimen. t Patient had a history of breast cancer and recent chemotherapy.
Outcome Well at 4 Well at 2 Did well Well at 4 Well at 4 Well at 6
y y mo mo rno
= present report; Ket = ketoconazole.
Patient no. [reference]
Sex/age (y)
1 [22] 2 [22] 3 [23] 4 [24] 5 [25] 6 [25]
F/58 M/65 M/54 M/49 M/43
M/49
Risk factor
Positive culture
Primary treatment
Recurrence
Secondary treatment
Gardening Farmer None Tree cutter Farmer Alcoholism
Yes Yes Yes Yes Yes Yes
Surgery, KI Surgery Surgery, KI Surgery, KI Surgery Surgery
Yes Yes Yes Yes Yes Yes
Surgery, KI AmB (943 mg) AmB (450 mg) AmB (3.8 g) AmB (1 g) Surgery, AmB (1.5 g)
Outcome Well Well Well Well Well Well
at at at at at at
10 y 7 mo 6w 2y 4y 4y
NOTE. KI = potassium iodide; AmB = amphotericin B.
Table 3. Characteristics of nine patients with tuberculous CTS. Patient no. [reference]
Sex/ age (y)
Positive skin test
1 [38]
M/57
Yes
2 [39]
M/44
Yes
3 [40] 4 [40]
M/66
M/66
ND ND
5 [41]
F/46
Yes
Miliary tuberculosis, + gastric aspirate Hilar adenopathy, axillary node + for acid-fast bacilli Tuberculosis in 1940 Tuberculosis of the spine, age 18 No
6 [5]
M/50
Yes
No
7 [5]
M/66
Yes
Old perihilar scarring
8 [42] 9 [43]
M/41 F/72
No No
No No
Evidence of systemic disease
Other positive tests *
Primary treatment
AFS Culture Culture
Surgery, INH, PAS, STM Surgery, INH, RMP
No
None
Did well
No
None
Well at 7 w
Culture Culture
Surgery, INH, RMP Surgery, INH, EMB
No No
None None
Well at 1 y Well at 6 mo
Culture
Surgery, INH, RMP, EMB Surgery, INH, RMP, EMB Surgery, INH, EMB
No
None
Well at 12 mo
No
None
Fusion of wrist
No
None
Surgery Surgery
Yes Yes
Surgery, INH, RMP Surgery, chemotherapy
Flexion deformity of wrist Did well Did well
AFS Culture AFS Culture Culture Culture
NOTE. AFS = acid-fast stain; INH = isoniazid; PAS = p-aminosalicylic acid; STM * Refers to culture and histopathologic examination of surgical tissue specimen.
culous arthritis [32], and eight other cases of tuberculous arthritis are mentioned in a report on a large series of cases ofCTS [6,9, 10,33,34]. Specific details regarding the cases are not given in these reports. Three reports from the foreignlanguage literature also mention CTS due to infection with
Recurrence Secondary treatment
Outcome
= streptomycin; RMP = rifampin; ND = note done; EMB = ethambutol.
M. tuberculosis [35-37]. The remaining nine cases are summarized in table 3. It is believed that peripheral tuberculous arthritis results from reactivation of a latent focus of infection rather than from direct inoculation of the organisms [32]. Moreover, eight of
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Table 2. Characteristics of six patients with CTS due to S. schenckii.
Mascola and Rickman
914
responded to treatment with isoniazid, ethambutol, and rifampin. Treatment in the majority of these cases consisted of surgery and the administration of chemotherapy. Patients generally did quite well and experienced complete resolution of symptoms. Only three of 22 patients were experiencing mild residual functional deficits at follow-up. Since chemotherapeutic combinations varied according to the infecting species of Mycobacterium, the reader is referred to the original references for details regarding specific treatment regimens. Bacterial causes ofCTS. There have been eight reported cases of acute pyogenic hand infections that resulted in CTS (table 4). Etiologic organisms include Staphylococcus aureus, coagulase-negative staphylococci, t1-hemolytic streptococci, Enterococcus faecalis, and Clostridium histolyticum. In marked contrast to patients with CTS caused by fungal or mycobacterial infection, all eight of these patients experienced some residual functional or sensory deficit after treatment. Not included in table 4 are three cases of CTS secondary to infection with Neisseria gonorrhoeae [65-67]. One patient had a clinical syndrome consistent with disseminated gonorrhea; however, on presentation, two others were asymptomatic except for the symptoms of CTS. Cultures of surgical specimens in both cases yielded N. gonorrhoeae. Disseminated gonococcal infection that manifests solely as monoarticular disease has been reported previously [68]. Borrelia burgdorferi. Infection with B. burgdorferi has recently been reported to cause CTS [68]. In a prospective study of patients with late-stage Lyme disease, Halperin et al. [69] found that 19 of 76 patients had symptoms of intermittent hand paresthesias, including 14 who had neurophysiologic evidence of entrapment of the median nerve at the wrist. All patients had been referred to these authors for evaluation of systemic or rheumatologic problems; in none of these cases were the neurologic symptoms the patient's primary problem. Clinical evidence of hand tenosynovitis or arthritis was present in two of the patients. Thus, mild cases of CTS may be fairly common in patients who have late-stage Lyme disease. Finally, toxic shock syndrome has also been associated with
Table 4. Characteristics of eight patients with bacterial CTS. Patient no. [reference]
1 [60] 2 [60] 3 [61] 4 [61] 5 [61] 6 [62] 7 [63] 8 [64]
Sexl age (y)
Trauma to hand
Culture results
Treatment
Outcome
F/58 F/53 F/68 M/52 F/50 F/IO M/23 M/24
Yes Yes No Yes Yes Yes No Yes
CN-Staph Sterile pus p-strep Polymicrobial * Sterile exudate Staphylococcus aureus Clostridium histolyticum S. aureus
Surgery, antibiotics Surgery, antibiotics Surgery, antibiotics Surgery, antibiotics .Surgery, antibiotics Surgery, antibiotics Surgery, antibiotics Aspiration, antibiotics
Mild motor and sensory deficits Persistent paresthesias Mild sensory deficit Mild functional and sensory deficits Slight sensory deficit Mild sensory deficit Mild functional impairment Significant median nerve palsy
NOTE. eN-Staph = coagulase-negative staphylococci; l3-strep = l3-hemolytic streptococci. * S. aureus; Enterococcus faecalis; l3-hemolytic streptococci.
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nine patients who are described in table 3 had a history of a reactive tuberculin test or an abnormal chest roentgenogram. Evidence of active disease was noted for two of these patients. Treatment of peripheral tuberculous arthritis has generally consisted of surgery and the administration of antituberculous medications, with good results [32,40]. In this review, seven of nine patients who were treated surgically and received chemotherapy did well. Two patients underwent surgery and did not receive medical treatment; both experienced recurrent episodes of CTS. Other Mycobacterium species. In reviewing the literature, we found 23 reported cases of CTS due to nontuberculous mycobacteria. The species implicated in these cases included Mycobacterium marinum (7 cases) [44-46]; the Mycobacterium avium complex (5 cases) [47-49]; Mycobacterium kansasii (4 cases) [47, 50-52]; Mycobacterium chelonei [53]; Mycobacterium fortuitum [14]; Mycobacterium szulgai [16]; Mycobacterium terrae [54]; Mycobacterium malmoense [55]; Mycobacterium leprae [56]; and Mycobacterium bovis [57]. Most cases occurred in patients with a history of exposure to the organism (e.g., contaminated water or fish infected with M. marinum) or a history of minor trauma to the hand. Only two patients had an underlying immunocompromising condition. Several of these cases deserve comment. The patient with CTS secondary to infection with M. leprae had no cutaneous manifestations of leprosy. In this case the diagnosis was made by visualization of acid-fast organisms that were isolated from a biopsy specimen of the median nerve, on the basis of a positive lepromin test, and on the basis of clinical response to dapsone. M. leprae is the only mycobacterium that specifically affects the peripheral nerves, and involvement of the median nerve is a common manifestation of leprosy [56]. However, this rarely occurs in the absence of ulnar neuropathy [56-59], and therefore this case of CTS represents a rare manifestation of leprosy. The M. bovis infection occurred in a patient who stuck herselfwith a Pasteur pipette that contained the Calmette-Guerin bacillus. CTS developed several weeks later and the patient
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RID 1991;13 (September-October)
Infectious Causes of CTS
Summary We have reviewed the known infectious causes of CTS, including bacteria, mycobacteria, fungi, viruses, and parasites. These are summarized in table 5. In most of the cases described, an infectious etiology was not suspected until it became apparent incidentally on surgical exploration or carpal tunnel release. The pathogenesis of CTS was not clear in all cases. Some organisms directly infect the tissues of the carpal tunnel, multiply, and compress the median nerve. Infection may occur via hematogenous spread (N. gonorrhoeae) or direct inoculation (sporotrichosis, M. marinum). In systemic illnesses such as rubella and toxic shock syndrome, it is possible that CTS results from an immune response to in-
Table 5.
Summary of infectious causes of CTS.
Cause of CTS
Histoplasma capsulatum Sporothrix schenckii Mycobacterium tuberculosis MOTT Pyogenic bacteria* Neisseria gonorrhoeae Borrelia burgdorferi TSS Rubella virus Rubella vaccine Guinea worm
No. of cases
Reference
6 6 26 23 8 4 14 4
[11-15] [22-25] [6, 9, to, 32-43] [16, 44-57] [60-64] [65-67] [69] [70] [77-82] [83-86] [89]
13 17 1
NOTE. MOTT = mycobacteria other than tuberculosis; TSS syndrome. * See text for specific organisms.
=
toxic shock
fection or the production of an exotoxin. Bilateral CTS is more common in patients with these systemic illnesses. Although the exact prevalenceof infectious CTS is unknown, infection should be considered in cases of prolonged or recurrent CTS. If symptoms are severe enough to warrant carpal tunnel release, tissue specimens should be obtained for microscopic evaluation and culture. Most patients with infectious CTS respond well to appropriate chemotherapy or surgery plus chemotherapy. Acknowledgment The authors thank Judy Emmons for assistance in the preparation of this manuscript.
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cases of CTS. Sahs et al. [70] reported four patients with toxic shock syndrome who experienced bilateral CTS during their illness. Symptoms resolved slowly in all patients. Although neuromuscular sequelae have been described in patients with toxic shock syndrome [71], the pathogenesis of CTS in patients with this disorder is not known. Rubella-induced CTS. Arthritis is a relatively common complication of rubella that occurs most often in young women. The hands, wrists, and knees are affected most frequently [72]. In 1940 several letters appeared in the British Medical Journal that described pain, numbness, and weakness of the hands of women with rubella [73-76]. Although these cases were not described in enough detail to document the presence of CTS, there have been 13 subsequent reports of cases of CTS associated with rubella [77-82] and 17 cases have occurred following immunization against rubella [83-86] (13 adults and 17 children). All cases that occurred in adults were in young women and the symptoms almost always occurred bilaterally. In children the symptoms were usually bilateral as well, but in contrast to adults, both sexes were affected. In only one instance did a patient undergo surgery for carpal tunnel release. Symptoms resolved over several weeks in all cases. Hepatitis B virus. Massey et a1. [87] described a 29-yearold woman who had a protracted hepatitis B virus infection and right-sided CTS. However, CTS did not develop until the sixth month of illness and there were no other signs of arthritis. Therefore, in this case we cannot be sure that CTS was related to the hepatitis infection. Guinea worm infestation. The guinea worm, or Dracunculus medinensis, is acquired by drinking water that contains crustaceans which have ingested the larvae of this nematode [88]. A report from Madras, India, described a 55-year-old woman with guinea worm infestation of the right wrist that caused CTS [89]. At surgical exploration, a calcified guinea worm was observed among the flexor tendons, adhering to the median nerve. After the parasite was removed the patient did well.
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RID 1991;13 (September-October)
RID 1991;13 (September-October)
Infectious Causes of CTS
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