Actinomycetoma is a chronic granulomatous infection of the subcutaneous tissue caused by fungus-like bacteria, that is, Nocardia sp, Actinomyces sp. This infection is common in tropical countries like India, though the disease is worldwide in distribution. A 22-year-old female who presented with multiple fistulas, sinus tracts, and pain and swelling over left foot following trauma 6 years back. A diagnosis of actinomycetoma was made based on clinical and microbiological ground. Two years after amputation there was recurrence and finally took multiple antibiotic therapies but was left without cure. Another case was that of a 37- year-old male who presented with multiple fistulas, sinus tracts, and pain and swelling on the right sole 6 months after prick of bamboo stick. There was a recurrence after amputation. Both the patients were treated with a Wallace regimen (Amikacin with Cotrimoxazole/Minocycline/Ciprofloxacin). Patient showed excellent response with healing of all sinuses after 2 months of therapy. Keywords: Actinomycetoma, amputation, grain
An actinomycetoma is a mycetoma caused by filamentous bacteria like Nocardia asteroides, Streptomyces somaliensis, and Actinomadura madurae.[1] They produce pyogenic lesions with formation of interconnecting sinus tracts that contain sulfur granules. [2] In many cases this soil fungus implants into the subcutaneous tissue by minor trauma, [3] grows slowly and extends to the skin and form skin sinuses, which drain granules. [4] Actinomycetoma requires amputation when complication sets in. The incidence of Nocardial mycetoma in Indian reports varies from 5.2% to 35%. We report this case because there are few reported case of post amputation recurrence of actinomycetoma in Kolkata. [5]
The first case was a 22-year-old female who presented with multiple fistulas, sinus tracts, and pain and swelling over left foot following trauma 6 years back for which she underwent amputation and took multiple antibiotic therapies but was left without cure. Two years after amputation she came with same symptoms and signs. Discharged grain was soft, small, and yellow-whitish. Grains were stained both by gram stain and modified Z-N stain. They were Gram-positive and acid-fast, respectively. Granules were cultured on Saburaud’s dextrose agar and incubated at 35-37°C aerobically for 3-14 days, Nocardia species produced orange to pink, waxy folded colonies covered with white aerial hyphae. They were catalase positive and urease positive. This patient was ultimately cured with 2 years thorough treatment with Amikacin with Cotrimoxazole/Minocycline/Ciprofloxacin). The second case was a 37-year-old male who presented with multiple fistulas, sinus tracts, and pain and swelling on the right sole 6 months after prick of bamboo stick. He was from a rural area. He was a farmer and remembered a history of occupational blunt trauma to his sole 6 months back. There was no history of diabetes mellitus or other causes of persistent wound infection. Physical examination revealed normal vital signs, but had a tender swollen right sided sole with multiple discharging sinuses. The laboratory results were unremarkable. Routine hemogram was within normal limit. The microbiological cultures of soft tissue for aerobic and anaerobic organisms showed growth of A. madurae. Colony in Saboraud’s dextrose agar had a glabrous, waxy, membranous or mucoid, heaped, and folded appearance. Gram staining showed Gram-positive bacilli, confirming the diagnosis of actinomycosis. Treatment was started with high doses of penicillin and was continued for 9 months. But there was no recovery. Below knee amputation was done 10 months after unsuccessful treatment. But within 3 years there was recurrence of A. madurae infection over thigh close to the amputation stump. He was successfully treated by combination of antibiotics like Amikacin with Cotrimoxazole/Minocycline/Ciprofloxacin.
Actinomycetoma is caused by actinomycetes which include the genera Nocardia, Actinomyces, and Streptomyces. The members of the genus Nocardia are filamentous Gram-positive bacteria that are ubiquitous soil saprophytes. N. Brasiliensis is the main pathogenic organism followed by N. Asteroides, which usually causes fulminant systemic infection. They enter into the skin through traumatic inoculation. Nocardia is detected by direct smears from such specimens typically show Gram-positive, beaded, branching filaments, which are usually acid fast stained by modified Kinyoun stain. Standard blood culture media support the growth of Nocardia organisms, but prolonged incubation (up to 2 weeks) and blind subcultures may be required for their detection. The identification of characteristic “sulfur granules” or “grains” which contain the infectious organisms, confirms the diagnosis of type of mycetoma. Grains from eumycetomas are larger and black or white-colored seen with the naked eye. Actinomycetoma grains are white to yellow in color, not seen with naked eye. Previously a case of actionomycosis of the amputation stump, with a possible underlying chronic actinomycotic osteomyelitis of the left tibia was reported by Sharma et al. Recurrence of post amputation actinomycetoma can be reduced if operation is done under appropriate antibiotic cover, that is, (Amikacin with Cotrimoxazole/Minocycline/Ciprofloxacin) Wallace regimen.
We acknowledge all the laboratory technicians of both medical colleges who helped us to finish this work.
Source of Support: None, Conflict of Interest: None
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