A rare case report of cerebral phaeohyphomycosis mimicking glioma in a patient of rheumatic valvular heart disease

Abstract

Phaeohyphomycosis is a collective term used for fungal infections caused by moulds and yeasts that have brown pigmented cell walls due to the presence of melanin. These are also known as dematiaceous fungi. We report this patient who presented with headache, right hemiparesis, slurred speech, and altered sensorium. Patient was a known case of rheumatic valvular heart disease (RVHD) and had undergone balloon valvotomy for mitral stenosis 1 year back. Radiological features were suggestive of high grade glioma. Left fronto-parietal decompressive craniectomy with complete excision of mass lesion was performed. Histopathological examination of the surgical specimen revealed multiple granulomas with giant cells. These giant cells contained branched septate pigmented fungal hyphae in their cytoplasm. After the histopathology report, patient was started on intravenous amphotericin and was discharged on oral itraconazole 200 mg twice daily. Unfortunately, the patient was non-compliant and stopped taking oral itraconazole after 1 month. He landed up in fulminant fungal meningo-encephalitis and died 10 weeks after the initial diagnosis. We report a rare case of cerebral phaeohyphomycosis in a patient of RVHD which, to our knowledge, is nowhere mentioned in the literature.

Cerebral phaeohyphomycosis: A rare case from central India

“…[2] Contiguous spread through paranasal sinuses or trauma resulting into implantation of fungal elements into the skin and its subsequent dissemination may be the cause of disease. [5] However, in our case, the patient neither had sinusitis nor did he present any history of trauma, unlike the reports presented by few authors. Several studies have also correlated the cause of infection with the patient's occupation.…”

“…Only four species like C. bantiana , Exophiala dermatitidis , Ramichloridium mackenziei and Ochroconis gallopavum are known to have a high neurotropic potential. [ 1 4 ] C. bantiana accounts for the majority of cases documented till date[ 5 6 ] and in our case also the etiological agent detected was C. bantiana . The fungus is known by several names such as Cladosporium trichoides , X. bantiana , X. emmonsii and Cladosporium bantianum .…”

“…Several studies have also correlated the cause of infection with the patient's occupation. [ 1 3 5 ] Though, the life-threatening fungal infections are often associated with severe immuno-compromised status, a typical but confounding and peculiar feature of this infection is that, it is increasingly recognized in immuno-competent persons with no underlying diseases. [ 1 2 9 ] The disease has also been frequently reported in males and in our case also the patient was an immuno-competent male.…”

“…[ 1 2 9 ] The disease has also been frequently reported in males and in our case also the patient was an immuno-competent male. [ 1 2 3 5 9 ]…”

“…Radiological investigations initially provided the extent of spread and later on the evidences of resolution so that the anti-fungal treatments could be individualized. [ 5 ] Treatments have largely been unsuccessful in most of the cases of cerebral phaeohyphomycosis. It must be individualized with surgical resection and optimized anti-fungal therapy.…”