Entomopthoromycosis-Report of four cases

Introduction

Entomopthoromycosis is a rare sporadic subcutaneous infection that is largely restricted to tropical areas of Africa, Asia and South America. Neither of the two clinically distinct forms of this mycosis occurs preferentially in patients with underlying disease or defective immunity.

Subcutaneous zygomycosis caused by Basidiobolus ranarum manifests clinically as a firm painless disciform nodule on the trunk or extremities. If untreated the nodule may enlarge and spread locally but systemic dissemination is extremely uncommon. The infection is probably initiated by penetrating trauma.

Rhinofacial zygomycosis caused by Conidiobolus coronatus is a locally progressive infection of the nasal cavity, paranasal sinuses and soft tissues of the face. The microscopic features of both forms of entomopthoromycosis are similar and give rise to an eosinophilic granuloma situated in the subcutaneous tissues. Here we report four cases of entomopthoromycosis. [Figure - 1]

Case Report

Case - 1. A 56-year-old man came with erythematous plaques with crusting over the face, trunk and lower extremities. The general health was good. [Figure - 2]

Case - 2. A 45-year-old man came with history of skin lesions on and off since 3 months. On examination there were large erythematous plaques with erosions and crusting on the trunk and the lower extremity.

Case - 3. A 35-year -old man came with erythematous papules and plaques over the face, depressed nose, nodules an extremities with discharging sinuses and lichenified lesions on the lower limbs. This patient was a known case of leprosy.

Case-4. A 45-year-old man came with history of nodules on the extremity and trunk.

In all the four cases there was no history of trauma and the patients′ general health was good.

Discussion

Entomopthoromycosis is a potentially curable disease which can masquerade as a neoplasm.[1] It has been known as a saprophytic fungus in plant debris and soil and is of low virulence. The mode of infection is not known but it is assumed that traumatic implantation may play a role or it is possibly transmitted by infected insects. It is a rare subcutaneous infection seen in tropical areas of Africa and Asia.[1] It manifests in two clinically distinct forms - subcutaneous zygomycosis and rhinofacial zygomycosis.

The commonest presentation is painless lesions on the extremities and the trunk.[2],[3] In addition Case no 1 and 3 had lesions on the face as well.[1],[4] Isolated facial involvementwas not seen. All the patients were immunologically competent and HIV status was negative in all cases.[5] Only one patient had underlying disease (Known case of lepromatous leprosy). AFB was negative in this case. Until now no case of entomopthoromycosis occurring in a patient of Hansen′s has been reported in literature. Their association is not known.

Biopsy of the lesion shows multiple granulomas, foreign body giant cells and eosinophils. In the midst of eosinophilic Splendore Hoepple[6] material non septate fungal hyphae are seen.[2],[5] Special stains, periodic acid schiff and silver methanamine are positive.