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DOI: 10.1016/j.jhsb.2003.09.011 © 2004 The British Society for Surgery of the Hand
Mycetoma of the Hand Caused by Gordona Terrae: A Case ReportFrom the Departments of Plastic Surgery and Tropical Medicine, University Medical Centre Nijmegen, Nijmegen, The Netherlands Correspondence: Dr X.R. Bakker, Department of Plastic Surgery, University Medical Centre Nijmegen, P.O. Box 9101, 6500 HB Nijmegen, The Netherlands. Tel.: +31-24-3615242; fax: +31-24-3619593; E-mail: xrbakker{at}planet.nl
Key Words: mycetoma • actinomycetoma • actinomyces • Gordona terrae • hand surgery
Mycetoma is a granulomatous, infectious, non-contagious disease of the subcutaneous tissues, which is mainly seen around the equator between latitudes 15°S and 30°N. It is endemic in areas of India, Pakistan, Indonesia, Central (Mexico) and South America (Brazil, Colombia and Venezuela), Senegal, Somalia and Sudan. Usually, the infection occurs after a minor injury such as a thorn prick and it mainly affects men aged 20 to 40 years. The causative agents are found in soils and on woody plants. The disease can be caused by either true fungi (eumycetoma) or aerobic actinomycetes (actinomycetoma) (McGinnis, 1996). In 70% to 80% of cases the infection is on the foot, because people in the endemic areas are often barefoot. The hand is involved in approximately 4% to 7% of cases (Mahe et al., 1996; Tautenhahn et al., 1977). After the minor injury a silent period takes place. Months to years thereafter, a subcutaneous, painless nodule develops at the site of injury. This explains why the patient often does not remember a history of trauma. The lesion grows slowly and extends to the skin, where skin sinuses form with drainage of grains containing clusters of fungi or bacteria. The triad of subcutaneous nodule, sinus formation and discharge of grains is specific for mycetoma (Fahal and Hassan, 1992). Secondary infections are painful and often produce purulent debris. The sinuses usually heal when the grains are excreted, but after a period of time new sinuses develop beside the old ones. In the late stages adjacent organs and bones can be affected, causing organ failure, fractures and osteomyelitis. Lymphatic or haematogenic spread is uncommon and the disease is not lethal, except when the tumour penetrates areas such as the extradural space in a case of craniofacial mycetoma (Baril et al., 1999). Many causative agents for mycetoma are known. For eumycetoma 31 different fungi have been documented (McGinnis, 1996). For actinomycetoma six bacteria have been found so far (Warren, 1996).
In 1998, a 15-year-old boy from Sierra Leone pricked the base of his left thumb with a thorn while in the jungle. A few months later an inflammation developed in the thenar region and pus, blood and grains discharged from sinuses which then healed. Every few weeks a new inflammation appeared a few millimetres away from the old lesion. The patient was presented to the Tropical Medicine Clinic of the University Medical Centre Nijmegen in 2001, with the clinical suspicion of mycetoma. On examination there was a 7 cm diameter tumour which was elevated 1.5 cm above the skin surface. There were multiple sinuses excreting granular material (Fig 1). Thumb function was virtually normal, but the conspicuous tumour with discharging sinuses interfered with the patient's social and professional life.
Incision biopsy showed a non-specific inflammation and adjuvant stains (Gram, Periodic Acid Schiff, Grocott, Giemsa, Ziehl-Neelsen, Spirochetes) were all negative. Bacteriological cultures were also negative. Blood tests showed no signs of infection, nor was there any suspicion of tuberculosis. No bone abnormality was seen on radiological examination of hand and wrist, but ultrasound revealed that the median nerve and flexor tendons were displaced by the tumour.
Operation
Microbiology and histology Samples were incubated aerobically on agar templates at 35°C. After 7 days a Gram-positive, coccoid, weakly acid-fast bacterium was isolated. Adjuvant electrophoresis by ribosomal DNA hybridization techniques matched an actinomyces species identified as Gordona terrae. Antimicrobial sensitivity testing revealed that the organism was sensitive to doxycycline. Histological examination of the complete specimen showed microbacterial colonies matching mycetoma (stains: Gram-positive, Periodic Acid Schiff-positive, Grocott-negative).
Although much is known of the cause of mycetoma, controversy exists as to its treatment. It is known that surgical treatment of mycetoma leads to a recurrence rate of 20% to 90% (Fahal and Hassan, 1992), probably because of attempts to preserve function. A distinction should be made between eumycetoma caused by fungi and actinomycetoma caused by bacteria, the aerobic actinomycetes. In eumycetoma, surgery aims to reduce the bulk of tumour. Although good results with only surgery in eumycetoma have been described (Visvanathan, 1989), adjuvant treatment with antimycotics is recommended (McGinnis, 1996; Smith and Kutbi, 1997). However, good results are achieved with conservative treatment, using only antimycotics such as ketoconazole (Venugopal and Venugopal, 1993). The success of treatment is dose-dependent (Mahgoub and Gumaa, 1984) and patients must be treated for months to years, with the risk of potentially serious side effects. The rate of cure in eumycetoma is less than 50% (McGinnis, 1996) and it results in a more chronic disease than actinomycetoma. Actinomycetoma can be cured in 63% to 95% of the cases (Fahal and Hassan, 1992; Welsh, 1993) and a combination of antibiotics is recommended to avoid drug resistance. Surgery is indicated if the bacteria are resistant to antibiotics or if the diagnosis is uncertain. Whether surgery combined with antibiotics or the use of antibiotics alone is the treatment of choice depends often on local circumstances and medical facilities. Identifying the causative organism in mycetoma is often difficult. Most fungi and actinomyces species grow slowly on standard culture media (De Hoog et al., 1993) and often the causative organism remains unidentified. As an incision biopsy did not reveal the diagnosis in our case, we decided to excise the tumour and, once the causative agent was known, the patient received adjuvant antibiotics. These were only given for 2 weeks because the bulk of tumour had been removed macroscopically. Normally, recurrences are seen within 6 to 9 months (Tautenhahn et al., 1977) and at 1 year followup the patient is still free of disease. Gordona terrae is a rare actinomycetes species. In humans, only four patients have been described with an infection caused by Gordona terrae and three of these were immunocompromized (Buchman et al., 1992; Drancourt et al., 1994, 1997).
The authors would like to thank M. Wulf from the Department of Microbiology of the University Medical Centre Nijmegen for finding the causative agent by microbiological techniques. Received for publication April 16, 2003. Accepted for publication September 19, 2003.
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