Eur J Plast Surg (2004) 27:143–145 DOI 10.1007/s00238-004-0636-8
CASE REPORT
N. Sungur · A. Uysal · M. Gms¸ · U. Koer · M. aydere · F. S¸ahin
Combined ulnar and median nerve compression due to atypical mycobacterial tenosynovitis: case report Received: 14 July 2003 / Accepted: 13 March 2004 / Published online: 8 June 2004 Springer-Verlag 2004
Abstract A 39-year-old woman with signs, symptoms and laboratory findings of carpal tunnel syndrome and ulnar compression is presented. Radical surgical debridement, excision of the affected synovium and early postoperative mobilization were performed. Mycobacterium marinum was identified from the culture of the excised flexor tenosynovium. Neither pulmonary nor extrapulmonary tuberculosis was diagnosed by X-ray and the other laboratory examinations. There has not been any previously reported case of mycobacterial tenosynovitis causing ulnar and median nerve compression together.
Introduction Since the introduction and extensive use of effective antibiotics for the treatment of tuberculosis in 1940’s, the incidence of mycobacterial infections of the extremities has decreased [12]. Infections of the hand due to mycobacteria have been even rarer, especially after the use of specific chemotherapeutic agents [1, 18]. Mycobacterial infections of the hand usually occur in the tenosynovium, and may cause ruptures of the tendons, or may disseminate to the joints or bones, if not treated. The enlarging mass may cause sensory or motor deficiency by compression [2]. Currently, atypical mycobacteria are the primary cause of hand infections, although mycobacterium tuberculosis was responsible more often in the past [19]. Mycobacterium marinum and mycobacterium kanN. Sungur ()) · A. Uysal · M. Gms¸ · U. Koer Department of Plastic and Reconstructive Surgery, Ankara Training and Research Hospital, Ankara, Turkey e-mail:
[email protected] N. Sungur Glseren sokak 15/7, Maltepe, Ankara, Turkey M. aydere · F. S¸ahin Department of Pathology, Ankara Training and Research Hospital, Ankara, Turkey
sasii are the most common atypical bacteria that cause hand infections [5, 19]. Any mass lesion located in the carpal tunnel may cause carpal tunnel syndrome (CTS) [14]. Trauma, tumors and systemic diseases, especially endocrine and infectious, are reported to be apparent causes of carpal tunnel syndrome [14, 17]. Mycobacteria, histoplasma capsulatum, coccidioides immitis, pyogenic infections, sporothrix schenkii and rubella are the most common infectious agents [14]. These may show similar clinical, operative and histological findings [12]. Mycobacterial tenosynovitis is known to be a cause of carpal tunnel syndrome, but there are a limited number of reported cases and none of these case reports have demonstrated an accompanying ulnar nerve compression leading to sensory and motor deficiency [3, 4, 6, 8, 9, 10, 12, 13, 16, 18]. A unique case of ulnar nerve compression accompanying carpal tunnel syndrome due to mycobacterial tenosynovitis is reported.
Case report A 39-year-old woman presented with swelling of the distal part of her right forearm and the wrist. She had a history of a short period of hospitalization 1 year before with fever, cough and weight loss but no apparent diagnosis was achieved after a thorough evaluation, and she was discharged without any treatment. The swelling of her right wrist occurred soon after and slowly enlarged. The patient suffered from pain and occasional numbness of all fingers and a decreased range of motion of the fourth and fifth fingers, but she did not have the previous systemic problems. She also had a longstanding history of regular aquarium cleaning. Physical examination revealed a 641.5 cm mass on the volar side of the right distal forearm and the wrist. Tinel and Phalen tests were positive. Ulnar arterial pulsation was weakly palpable, probably due to the mass effect. Interosseous muscles were functioning normally. Atrophy of the thenar region and hypoesthesia of the thenar and hypothenar regions was noted. Hypoesthesia of the thumb, fourth and fifth fingers was apparent. Nerve conduction studies confirmed ulnar and median nerve compression. Routine blood counts and biochemical analysis were normal. Serological tests of rheumatoid and collagen tissue diseases were negative; chest and wrist X-rays showed no abnormalities. The forearm was explored under general anesthesia, using a tourniquet. After the incision of the fascia of the forearm, the large cystic mass surrounding the superficial and deep flexor tendons
144 became apparent. The median nerve was compressed and displaced radially and superficially. The ulnar nerve was also compressed and displaced to the ulnar side (Fig. 1). Median and ulnar neurolysis were performed as well as flexor tenosynovectomy. The excised material was sent for bacterial, mycobacterial and fungal cultures as well as histopathological examination. Active exercises were started on the postoperative day 5. Histopathological examination revealed granulomatous structures consisting of caseous necrosis at the center of the fibrous stroma, surrounded by lymphocytes and histiocytes peripherally and Langhans’ giant cells outside them (Fig. 2a, b). No acid-fast bacilli were detected; bacterial and fungal cultures were sterile. Mycobacterium marinum grew on the mycobacterial culture (Lowenstein-Jensen at 30C) 7 weeks later. The patient was referred to the chest and infectious diseases clinic for appropriate treatment. Apart from the positive PPD test, no abnormalities or systemic mycobacterial pathology was detected on further evaluation, and no pulmonary or extrapulmonary systemic tuberculosis was detected. INH, ethambutol, morfozinamid and rifampin combination was preferred for treatment. The patient is still being followed up. Fig. 1 Intraoperative view of the tumor. Displacement of the median and ulnar nerve due to compression. M, median nerve; U, ulnar nerve; L, lesion; CT, released carpal tunnel; FCU, flexor carpi ulnaris tendon; VCL, volar carpal ligament; " , compression and displacement
Fig. 2 a Caseous necrosis at the center of the granuloma (H&E 50). b Granulomas consisted of Langhans’ giant cells and epitheloid histiocytes are seen beneath the synovial epithelium, on histopathological examination (H&E 100)
Discussion The most common symptoms of mycobacterial infections of the hand and the wrist are painless, progressive swelling and joint stiffness [12]. Severe cases present with pain, paresthesia or weakness, but they are not common. Impairment of flexion or extension due to pain or compression may occur, as well as localized swelling and erythema [7, 9]. Systemic disease does not accompany infections of the hand or the wrist in most cases. Fever and sweating are also uncommon. Tenosynovitis is the most common form of mycobacterial infection of the hand and wrist. Flexor tendons at the wrist and the radioulnar bursa (the compound palmar ganglion) are mostly affected, and the digital flexor sheath and the distal wrist compartments are the least common [12, 18]. Bone and joint infections are almost always secondary to primary synovial disease [12]. The accurate diagnosis of mycobacterial tenosynovitis is time consuming [12, 18]. Non-specific or rheumatoid synovitis is usually considered before the diagnosis of mycobacterial disease [3]. Histological and bacteriological examination is essential for the diagnosis [15]. The absence of acid-fast bacilli on staining does not rule out mycobacterial infection [1, 2]. Definitive diagnosis relies on biopsy and culture of the infected bone or rice body [1, 11]. In the early stages of mycobacterial infections of the wrist and hand, swelling of the involved compartment with tenosynovitis and a serous exudate are observed. The inflamed synovium surrounds and compresses tendons and nerves without direct invasion, defined as the ‘hygromatous stage’ [12, 18]. As the pathology progresses, tendon sheaths become fibrotic, and adhesions occur, leading to joint stiffness. Fragmentation or rupture of the tendons, sinus formation and secondary infections are the late complications of severe cases [12]. Medical therapy is essential for the eradication of the infectious agent. Surgical intervention is indicated when medical therapy fails to cure the nerve compression or in the case of an abundant mass [4]. Persistent pain and sinus
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formation have been reported after ineffective medical treatment [2]. Corticosteroid injection is contraindicated, and previous steroid injection increases the necessity for surgical treatment [19]. Entrapments of a nerve or a decrease in the range of motion of the wrist or the fingers, due to compression of the mass, are indications for early surgical excision [2]. Early aggressive debridement may obviate the need for arthrodesis or amputation because of the aggressive nature of the organism and failure of conservative therapy to eradicate the infection [11]. Previous studies on mycobacterial infections of the hand and the wrist confirm the need for aggressive surgical debridement, excision of the affected synovia, early postoperative mobilization and antimycobacterial therapy [1, 2, 3, 10, 12]. The duration of the treatment is uncertain, but 6 to 12 months are recommended, depending on complete resolution of the lesions [2]. Long-term follow up is essential [15]. Differential diagnoses include sarcoidosis, foreign bodies, Crohn’s disease, brucellosis and fungal infections [2]. Our patient had a large mass in the distal forearm and the wrist which caused signs and symptoms of compression of the median and the ulnar nerves together. The enlarging mass due to tenosynovitis is known to cause sensory or motor deficits by compressing the adjacent nerves [2]. Mycobacterial tenosynovitis is known to be a cause of carpal tunnel syndrome, but reported cases are few and none of these case reports have demonstrated an accompanying ulnar nerve compression leading to sensory and motor deficiency [3, 4, 6, 8, 9, 10, 12, 13, 18]. A unique case of ulnar nerve compression accompanying carpal tunnel syndrome due to mycobacterial tenosynovitis is reported.
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