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Interesting Cases

Rare presentation of actinomycetoma

Case Report
A.H. Fahal and H. E. Sheik
Mycetoma Research Centre,
University of Khartoum,

The patients; is a 36 year-old gentleman, farmer from Western Sudan. He presented to the Mycetoma Clinic at Soba University Hospital, Khartoum, Sudan with massive swelling of the left side of the back extending down to the gluteal region. The condition stared 18 years with a small painless swelling in the left lower part of his back at a site of trauma following a fall from a tree. It then gradually regressed in size. Two year later the swelling gradually stared to increase in size, sinuses discharging yellow grains then developed. He had no past history of bilharziasis or haematemesis. His social, family or drug history is not contributory to his condition.

On examination he looks unwell and slightly pale. Head and neck, chest and cardio-vascular, central nervous system examination are within normal. He has gynaecomastia of the left breast, which is tender and firm in consistency. Abdominal examination revealed multiple distended veins extending from the left hypochondrial region to the left iliac fossa. The spleen is grossly enlarged otherwise normal clinical findings.

Local examination of the back revealed huge mass involving the whole left side of back extending to the chest wall and down to the gluteal region. The mass was painless, firm in consistency with areas of hypopigmentation. The skin was tethered to the deep structures with multiple healed sinuses.

His full blood count and ESR are within normal. Abdominal ultrasound showed an enlarged spleen with normal texture, normal splenic vein and evidence of bilharzial periportal fibrosis. Intravenous urography showed normal function of both kidneys, the left ureter was deviated medically and erosion of the left 12 rib and the first five lumber vertebrae.  Abdominal CT scan showed a huge mass at the back pushing the left kidney and ureter medical with erosion of the first five lumber vertebrae. An incisional biopsy was taken under local anesthesia and it confirmed the diagnosis of actinomycetoma due to Streptomyces somaliensis. MRI examination showed huge soft tissue mass.

The patient was stared on streptomycin sulphate in a dose of 14 mg/kg daily for one month then on alternate days and diaminodiphenyl sulphone (dapsone) in a dose of 1.5 mg/kg twice daily and Co-trimoxazole in a dose of 14 mg/kg twice daily. His portal hypertension was treated conservatively.  At two months follow up he showed dramatic clinical improvement the mass had regressed in size, sinuses had healed and the skin became less tethered to the underline tissue.