Vascular Pythiosis in a Thalassemic Patient

Abstract

Pythium insidiosum is a fungus that causes disease in both animals and humans. Human pythiosis is an emerging disease in the tropical, subtropical, and temperate regions of the world, occurring in localized and systemic or vascular forms. Most patients with arterial pythiosis have an underlying hemoglobinopathy, such as thalassemia. A case is presented of a thalassemic horse stable worker who developed an ulcerative cutaneous lesion on the lower left leg followed by progressive ascending involvement of the arteries of that extremity with a necrotizing arteritis with aneurysm formation. P. insidiosum was not isolated from the ulcer by culture or wet potassium hydroxide preparations but was diagnosed by histopathologic study of a biopsy. P. insidiosum infection was quickly confirmed by immunoblot method, aiding in preoperative decision making. Many systemic antibiotics or antimycotics have not been effective in the treatment of systemic pythiosis, and radical surgical removal of all infected tissue is the only method to ensure patient survival. An orally administered saturated solution of potassium iodide, amphotericin B-oral solution, and terbinafine has succeeded only in the cutaneous form but had no favorable effect on vascular pythiosis. It is likely that immunotherapy, successfully used in animal pythiosis, may be beneficial in the treatment of human vascular pythiosis.