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Recurrent histoplasmosis of the wrist: A case report
Recurrent Histoplasmosis of the Wrist: A Case Report Steven B. Care, MD, Stephen H. Lacey, MD, Cleveland, OH Histop[asmosis of the extremities is rare. A case of recurrent histoplasmosis with a 10-year latency between initial presentation and clinical recurrence is reported. Prolonged antibiotic treatment and debridement of bony involvement led to clinical resolution of this fungal infection after a follow-up period of 20 months. (J Hand Surg 1998;23A:1112-1114. Copyright 9 1998 by the American Society for Surgery of the Hand.)
Disseminated histoplasmosis is rare, but its manifestations are protean. Lung involvement predominates in 90% of cases, but histoplasma may involve bone and soft tissues. Rarely, peripheral involvement dominates the presentation. We report a case of recurrent upper extremity histoplasmosis without clinical evidence of pulmonary involvement presenting 10 years following its initial manifestation.
Case Report A 35-year-old male landscaper presented with a 1-month history of progressive hand symptoms of night awakening, numbness, and tingling in the radial digits. Work aggravated these symptoms and produced paresthesias in the median nerve distribution. Examination revealed decreased 2-point discrimination and boggy thickening around the carpal tunnel volarly, consistent with flexor tenosynovitis. No swelling or symptoms were noted in the extensor compartments. Wrist radiographs revealed cystic bone changes (Fig. 1), but chest radiographs were normal. Electromyography and nerve conduction studies demonstrated median nerve corn-
From the Department of Orthopaedic Surgery, University Hospitals of Cleveland, Cleveland, OH. Received foE publication April 1, 1997; accepted in revised form June 9, 1998. No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article. Reprint requests: Steven B. Care, MD, West Idaho Orthopaedic Fracture Clinic, 206 E Elm St, Caldwell, ID 83605. Copyright 9 1998 by the American Society for Surgery of the Hand 0363-5023/98/23A06~002253.00/0
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The Journal of Hand Surgery
pression, and the patient underwent carpal tunnel release with concurrent flexor tenosynovectomy. Findings included red-brown proliferative tenosynovium enveloping the flexor tendons and adjacent involvement within a cortical fenestration in the central volar capitate. The fenestration in the capitate was debrided of granulomatous tissue that apparently originated from the flexor tenosynovium. Extensive curettage of the capitate was not done at this time. Histologic examination revealed chronic granulomatous inflammation with giant cells. Histoplasmin complement fixation testing demonstrated mycelial and yeast phase antibody titers of 1:8 and 1:16, respectively. Silver stains failed to disclose fungal elements, but culture of the proliferative tenosynovial tissue grew Histoplasma capsulatum and skin testing demonstrated a positive histoplasmin reaction. After surgery, the patient was treated with ketaconazole for 4 months. There was complete resolution of symptoms and swelling at the 6-month follow-up visit. Repeat radiographs showed no change in the capitate cyst. The patient returned to his occupation, working vigorously for several years without pain. As his business grew, he became less involved in manual labor and focused primarily on economic issues. He occasionally noted mild discomfort in his wrist, but did not seek medical attention. Ten years later, boggy swelling and pain recurred about the extensor side of the wrist that was associated with radiographic progression of the cystic capitate lesion (Fig. 2). Exploration through volar and dorsal approaches 1 week later revealed extensive red-brown proliferative synovial tissue resembling rheumatoid pannus wrapping around
The Journal of Hand Surgery/Vol. 23A No. 6 November 1998
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Figure 1. Radiograph of the wrist at initial presentation demonstrating cystic lesions of the capitate with sclerotic borders.
Figure 2. Radiographic appearance at the time of clinical recurrence with progression of the cystic changes in the capitate.
tendons of the second through fourth compartments dorsally and the flexor carpi radialis volarly (Fig. 3). Extensive tenosynovectomy was performed as well as thorough curettage of the capitare cyst. Histologic examination again noted extensive chronic granulomatous inflammation with giant cells in both of these locations. Despite similar histology, histoplasma did not grow on culture of the second specimens. However, after consultation with infectious disease experts, the patient was treated with intraconazole for 6 months. There was complete clinical resolution of all swelling and pain. Testing for human immunodeficiency virus was negative, and the patient demonstrated no evidence of immunocompromise. Twenty months following the revision surgery, the patient had full range of motion, no pain or tenderness, and no swelling or soft tissue thickening. X-
rays revealed no evidence of new osteolysis and apparent healing of the capitate cyst.
Discussion
Histoplasma was originally isolated from bone marrow but also infects the soft tissues and has presented clinically as tenosynovitis or carpal tunnel syndrome, t-4 As in our case, bony involvement usually accompanies inflamed tenosynovium, and bone infection may precede tenosynovial involvement. However, Perlman et al5 reported infectious tenosynovitis at the wrist occurring in the absence of radiographic bone involvement. Thus, it is uncertain whether soft tissue or bone involvement is primary. Wrist infection recurred in Perlman et al's 5 case following a latency of 1 year; similar latent periods have been reported between onset of pulmonary his-
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Care and Lacey / Recurrent Histoplasmosis of the Wrist
recurrence in our patient, as the bone cyst seen radiographically was not thoroughly debrided during the initial procedure. Small bone cysts are common in the carpus and are usually degenerative, but failure to appreciate the significance of such cysts in the presence of infectious tenosynovitis may have allowed fungal osteomyelitis to persist, resulting in recurrence in our patient. On the other hand, resection without antibiotic support is likely to fail, as histoplasma may remain latent in the lungs or elsewhere inthe bone marrow. Current antifungal agents are highly effective, but recommendations depend on host immune status and the clinical picture. Severe disseminated disease in immunocompromised hosts is difficult to eradicate and requires intravenous treatment with amphotericin B followed by life-long oral therapy. A 3- to 6-month oral course of itraconazole or ketaconazole is well-tolerated and effective for milder, more focal disease and for maintenance therapy. 6 Histoplasma infection of the extremity is rare, but enters the differential diagnosis of compressive neuropathy at the wrist, cystic bone lesions, and prominent tenosynovial proliferation, with or without immunocompromise. F i g u r e 3. Gross appearance at re-exploration with boggy,
red-brown tenosynovial tissue enveloping extensor tendons of the fourth compartment.
toplasmosis and the development of peripheral involvement, a The present case demonstrates a prolonged period of 10 years between initial presentation and obvious clinical recurrence, suggesting that histoplasma can remain latent for as long as a decade. The ability of histoplasma to reside in bone marrow necessitates prolonged antibiotic treatment following resection of grossly involved tissue. Debridement of cystic bone lesions is indicated and may have been the focus of the latent infection, allowing
References 1. Omer GE Jr, Lockwood RS, Travis LO. Histoplasmosis involving the carpal joint: a case report. J Bone Joint Surg 1963;45A:1699-1703. 2. strayer DS, Gutwein MB, Herbold D, Bresaller R. Histoplasmosis presenting as the carpal tunnel syndrome. Am J Surg 1981;141:286-288. 3. 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. 4. Eglseder WA. Carpal tunnel syndrome associated with histoplasmosis: a case report and literature review. Mil Med 1992;157:557-559. 5. Perlman R, Jubelirer RA, Schwarz J. Histoplasmosis of the common palmar tendon sheath. J Bone Joint Surg 1972; 54A:676-678. 6. Wheat J. Histoplasmosis: recognition and treatment. Clin Infect Dis 1994;19:$19-$27 (suppl 1).
Disseminated histoplasmosis is rare, but its manifestations are protean. Lung involvement predominates in 90% of cases, but histoplasma may involve bone and soft tissues. Rarely, peripheral involvement dominates the presentation. We report a case of recurrent upper extremity histoplasmosis without clinical evidence of pulmonary involvement presenting 10 years following its initial manifestation.
Case Report A 35-year-old male landscaper presented with a 1-month history of progressive hand symptoms of night awakening, numbness, and tingling in the radial digits. Work aggravated these symptoms and produced paresthesias in the median nerve distribution. Examination revealed decreased 2-point discrimination and boggy thickening around the carpal tunnel volarly, consistent with flexor tenosynovitis. No swelling or symptoms were noted in the extensor compartments. Wrist radiographs revealed cystic bone changes (Fig. 1), but chest radiographs were normal. Electromyography and nerve conduction studies demonstrated median nerve corn-
From the Department of Orthopaedic Surgery, University Hospitals of Cleveland, Cleveland, OH. Received foE publication April 1, 1997; accepted in revised form June 9, 1998. No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article. Reprint requests: Steven B. Care, MD, West Idaho Orthopaedic Fracture Clinic, 206 E Elm St, Caldwell, ID 83605. Copyright 9 1998 by the American Society for Surgery of the Hand 0363-5023/98/23A06~002253.00/0
1112
The Journal of Hand Surgery
pression, and the patient underwent carpal tunnel release with concurrent flexor tenosynovectomy. Findings included red-brown proliferative tenosynovium enveloping the flexor tendons and adjacent involvement within a cortical fenestration in the central volar capitate. The fenestration in the capitate was debrided of granulomatous tissue that apparently originated from the flexor tenosynovium. Extensive curettage of the capitate was not done at this time. Histologic examination revealed chronic granulomatous inflammation with giant cells. Histoplasmin complement fixation testing demonstrated mycelial and yeast phase antibody titers of 1:8 and 1:16, respectively. Silver stains failed to disclose fungal elements, but culture of the proliferative tenosynovial tissue grew Histoplasma capsulatum and skin testing demonstrated a positive histoplasmin reaction. After surgery, the patient was treated with ketaconazole for 4 months. There was complete resolution of symptoms and swelling at the 6-month follow-up visit. Repeat radiographs showed no change in the capitate cyst. The patient returned to his occupation, working vigorously for several years without pain. As his business grew, he became less involved in manual labor and focused primarily on economic issues. He occasionally noted mild discomfort in his wrist, but did not seek medical attention. Ten years later, boggy swelling and pain recurred about the extensor side of the wrist that was associated with radiographic progression of the cystic capitate lesion (Fig. 2). Exploration through volar and dorsal approaches 1 week later revealed extensive red-brown proliferative synovial tissue resembling rheumatoid pannus wrapping around
The Journal of Hand Surgery/Vol. 23A No. 6 November 1998
1113
Figure 1. Radiograph of the wrist at initial presentation demonstrating cystic lesions of the capitate with sclerotic borders.
Figure 2. Radiographic appearance at the time of clinical recurrence with progression of the cystic changes in the capitate.
tendons of the second through fourth compartments dorsally and the flexor carpi radialis volarly (Fig. 3). Extensive tenosynovectomy was performed as well as thorough curettage of the capitare cyst. Histologic examination again noted extensive chronic granulomatous inflammation with giant cells in both of these locations. Despite similar histology, histoplasma did not grow on culture of the second specimens. However, after consultation with infectious disease experts, the patient was treated with intraconazole for 6 months. There was complete clinical resolution of all swelling and pain. Testing for human immunodeficiency virus was negative, and the patient demonstrated no evidence of immunocompromise. Twenty months following the revision surgery, the patient had full range of motion, no pain or tenderness, and no swelling or soft tissue thickening. X-
rays revealed no evidence of new osteolysis and apparent healing of the capitate cyst.
Discussion
Histoplasma was originally isolated from bone marrow but also infects the soft tissues and has presented clinically as tenosynovitis or carpal tunnel syndrome, t-4 As in our case, bony involvement usually accompanies inflamed tenosynovium, and bone infection may precede tenosynovial involvement. However, Perlman et al5 reported infectious tenosynovitis at the wrist occurring in the absence of radiographic bone involvement. Thus, it is uncertain whether soft tissue or bone involvement is primary. Wrist infection recurred in Perlman et al's 5 case following a latency of 1 year; similar latent periods have been reported between onset of pulmonary his-
1114
Care and Lacey / Recurrent Histoplasmosis of the Wrist
recurrence in our patient, as the bone cyst seen radiographically was not thoroughly debrided during the initial procedure. Small bone cysts are common in the carpus and are usually degenerative, but failure to appreciate the significance of such cysts in the presence of infectious tenosynovitis may have allowed fungal osteomyelitis to persist, resulting in recurrence in our patient. On the other hand, resection without antibiotic support is likely to fail, as histoplasma may remain latent in the lungs or elsewhere inthe bone marrow. Current antifungal agents are highly effective, but recommendations depend on host immune status and the clinical picture. Severe disseminated disease in immunocompromised hosts is difficult to eradicate and requires intravenous treatment with amphotericin B followed by life-long oral therapy. A 3- to 6-month oral course of itraconazole or ketaconazole is well-tolerated and effective for milder, more focal disease and for maintenance therapy. 6 Histoplasma infection of the extremity is rare, but enters the differential diagnosis of compressive neuropathy at the wrist, cystic bone lesions, and prominent tenosynovial proliferation, with or without immunocompromise. F i g u r e 3. Gross appearance at re-exploration with boggy,
red-brown tenosynovial tissue enveloping extensor tendons of the fourth compartment.
toplasmosis and the development of peripheral involvement, a The present case demonstrates a prolonged period of 10 years between initial presentation and obvious clinical recurrence, suggesting that histoplasma can remain latent for as long as a decade. The ability of histoplasma to reside in bone marrow necessitates prolonged antibiotic treatment following resection of grossly involved tissue. Debridement of cystic bone lesions is indicated and may have been the focus of the latent infection, allowing
References 1. Omer GE Jr, Lockwood RS, Travis LO. Histoplasmosis involving the carpal joint: a case report. J Bone Joint Surg 1963;45A:1699-1703. 2. strayer DS, Gutwein MB, Herbold D, Bresaller R. Histoplasmosis presenting as the carpal tunnel syndrome. Am J Surg 1981;141:286-288. 3. 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. 4. Eglseder WA. Carpal tunnel syndrome associated with histoplasmosis: a case report and literature review. Mil Med 1992;157:557-559. 5. Perlman R, Jubelirer RA, Schwarz J. Histoplasmosis of the common palmar tendon sheath. J Bone Joint Surg 1972; 54A:676-678. 6. Wheat J. Histoplasmosis: recognition and treatment. Clin Infect Dis 1994;19:$19-$27 (suppl 1).