Subcutaneous Granulomatous Inflammation due to Basidiobolomycosis: Case Reports of 3 Patients in Buruli Ulcer Endemic Areas in Benin

Abstract

Background Basidiobolomycosis is a rare subcutaneous mycosis, which can be mistaken for several other diseases, such as soft tissue tumors, lymphoma, or Buruli ulcer in the preulcerative stage. Microbiological confirmation by PCR for Basidiobolus ranarum and culture yield the most specific diagnosis, yet they are not widely available in endemic areas and with varying sensitivity. A combination of histopathological findings, namely, granulomatous inflammation with giant cells, septate hyphal fragments, and the Splendore-Hoeppli phenomenon, can confirm basidiobolomycosis in patients presenting with painless, hard induration of soft tissue. Case Presentations We report on three patients misdiagnosed as suffering from Buruli ulcer, who did not respond to Buruli treatment. Histopathological review of the tissue sections from these patients suggests basidiobolomycosis. All patients had been lost to follow-up, and none received antifungal therapy. On visiting the patients at their homes, two were reported to have died of unknown causes. The third patient was found alive and well and had experienced local spontaneous healing. Conclusion Basidiobolomycosis is a rare subcutaneous fungal disease mimicking preulcerative Buruli ulcer. We stress the importance of the early recognition by clinicians and pathologists of this treatable disease, so patients can timely receive antifungal therapy.

Basidiobolomycosis in Togo: clinico-pathological study of a series of 12 presumed cases

“…Microbiological confirmation by PCR for B. ranarum and culture provides the most accurate diagnosis, but they are not often available in endemic areas and with varying sensitivity. A combination of histopathological findings, granulomatous inflammation with giant cells, septate hyphal fragments and the Splendore-Hoeppli phenomenon, may confirm Basidiobolomycosis in patients with painless induration of soft tissues [ 1 , 6 ].…”

“…Azole derivatives have emerged as the treatment of choice for phycomycoses. Among them, ketoconazole has been shown to be effective in Basidiobolomycosis [ 5 , 6 , 18 ]. This imidazole has been used successfully for doses ranging from 5 to 10 mg/kg/day [ 23 , 24 ].…”

Disseminated and ulcerative basidiobolomycosis simulating a buruli ulcer in an immunocompetent girl in Southern Benin

“…as well as Brun et al . reported cases of basidiobolomycosis in Benin in areas known to be endemic to Buruli ulcer [ 5 , 8 ]. Although culture wasn´t performed, our case was classified as a basidiobolomycosis based on epidemiological, clinical, biological and histopathological arguments and the efficacy of antifungal drug administered.…”

“…Like Buruli ulcer, basidiobolomycosis most commonly affects children between 4-10 years of age [ 3 , 10 ]. This can lead to a diagnosis delay and the evolution can sometimes be fatal [ 5 , 12 ]. In these cases, the histopathology makes it possible to make the diagnosis.…”