Disseminated basidiobolomycosis in an immunocompetent woman

Basidiobolomycosis is a chronic subcutaneous infection of the trunk and limbs due to Basidiobolus ranarum. The disease is well known in tropical areas, although recent cases of gastrointestinal basidiobolomycosis have also been reported in Arizona. We describe a young immunocompetent women who had presented with eosinophilia and lung infiltrates. She subsequently died, and diagnosis of disseminated basidiobolomycosis was made on the basis of histological features at autopsy.     

Gastrointestinal basidiobolomycosis. Morphologic findings in a cluster of six cases.

We describe the histopathologic features of 6 cases of gastrointestinal basidiobolomycosis examined at 4 Phoenix, AZ, area hospitals during the last 4 years. Resected stomach and intestinal specimens were characterized by marked mural thickening with fibrosis, prominent tissue eosinophil infiltration and palisading granulomatous inflammation around pale fungal hyphae. In 2 cases, there was colonic perforation. Basidiobolus ranarum hyphae (associated with spore-like spherules in 4 cases) were identified within tissue sections; the irregularly branched, thin-walled, occasionally septated hyphae were typically surrounded by a thick eosinophilic cuff (Splendore-Hoeppli phenomenon). Although the histologic features of B ranarum are well described in the skin and subcutaneous tissue, gastrointestinal involvement has presented considerable diagnostic difficulty. Before the occurrence of this cluster of cases, intra-abdominal B ranarum infection has been reported only rarely.     

Basidiobolomycosis: a rare case report

We report a rare case of basidiobolomycosis seen in an 11-year-old girl from North-Eastern part of India. She presented with complaints of bilateral nasal block and nasal discharge for seven-eight months. CT scan of sinuses revealed polypoidal mass in all the sinuses with extradural extension. The tissue biopsy examined histopathologically and microbiologically, revealed Basidiobolus ranarum.     

An unusual case of gastrointestinal basidiobolomycosis mimicking colon cancer; literature and review

Gastrointestinal basidiobolomycosis (GIB), a rare fungal infection associated with high mortality, has been reported worldwide mainly from tropical and subtropical regions of Asia, USA, and Latin America. The clinical manifestations are highly diverse and non-specific depending on the underlying disease, but fever, abdominal pain, weight loss, diarrhea, constipation and chills have been observed. There are no prominent risk factors for GIB but climatic conditions and life style are related to this infection in arid and semi-arid regions. Therefore timely diagnosis and early treatment is a challenge. Herein, we present an unusual case of gastrointestinal basidiobolomycosis in a 54-year-old male, initially misdiagnosed as colon cancer. After follow-up, no evidence of relapse and the patient was successfully cured by liposomal amphotericin B. In addition, the differential diagnosis and histopathological findings are discussed with a review of the literature.     

Immunodiffusion test for serodiagnosing subcutaneous zygomycosis.

Culture filtrate antigens of Basidiobolus ranarum and Conidiobolus coronatus were analyzed by immunodiffusion (ID) with homologous rabbit antisera. B. ranarum and C. coronatus were each found to have five specific antigens. Results of tests with heterologous antisera indicated that all of the species shared at least one antigen. ID tests incorporating the specific precipitin bands as references were developed for detection of basidiobolomycosis and conidiobolomycosis. These tests were performed with sera from humans and horses with proven basidiobolomycosis and conidiobolomycosis as well as with control sera from humans and animals with and without heterologous mycotic and oomycotic infections. Only sera from cases of basidiobolomycosis and conidiobolomycosis produced lines of identity with the reference precipitates of B. ranarum and C. coronatus, respectively. The ID tests were found to be completely sensitive and specific for determining the etiology of zygomycosis caused by these two species. In addition they appeared useful for monitoring resolution of the infections.     

Gastrointestinal histoplasmosis presenting as colonic pseudotumour

We report a case of gastrointestinal histoplasmosis in a 45-year-old HIV positive man who was misdiagnosed as a case of colonic cancer. The patient presented with low-grade fever, pain in lower abdomen, anorexia and weight loss of six months duration. On examination a lump in the left iliac fossa was detected. Colonoscopy revealed stricture and ulcerated growth in the sigmoid colon. Radiological investigations suggested malignant/inflammatory mass in the sigmoid colon with luminal compromise. Patient was operated and ulcerated tissue was sent for histopathological examination, which revealed numerous intracellular, 2-4 microm, oval, narrow-based budding yeast cells suggestive of Histoplasma capsulatum. Subsequently, the patient developed fluffy opacities on X-ray chest. Examination of sputum revealed presence of acid-fast bacilli and yeast forms of H. capsulatum. Patient was started on amphotericin B but died on the seventeenth postoperative day. The diagnosis of histoplasmosis was made retrospectively. Atypical presentation and rarity of the disease led to this diagnostic pitfall. To the best of our knowledge this is the first report of gastrointestinal histoplasmosis presenting as colonic pseudotumour from India.     

Subcutaneous Hyalohyphomycosis Caused by Colletotrichum gloeosporioides

The coelomycete Colletotrichum gloeosporioides was isolated in pure culture from subcutaneous nodules of the left forearm and elbow of a farmer after traumatic injury. To our knowledge, we report the first case involving this fungus as an etiological agent of subcutaneous infection. The in vitro inhibitory activities of amphotericin B, itraconazole, ketoconazole, miconazole, flucytosine, and fluconazole were studied.     

Lung infection due to opportunistic fungus, Phialemonium obovatum, in a bone marrow transplant recipient: an emerging infection with fungemia and Crohn disease-like involvement of the gastrointestinal tract

We report the first case of Phialemonium obovatum fungemia with subsequent caseating granulomatas in the lung and Crohn disease-like involvement of the gastrointestinal tract in a bone marrow transplant recipient. This phaeoid fungus has been rarely described as an opportunistic infection in immunosuppressed patients. The patient was diagnosed with chronic myelogenous leukemia and underwent subsequent peripheral bone marrow transplant. After 6 months, he developed graft-versus-host disease of the skin and liver with fever and severe diarrhea. Fecal bacterial cultures and cytomegalovirus serologies were negative. Computed tomographic scan showed a peripheral pulmonary mass. A lung wedge biopsy of the lesion showed septate branching hyphae (4-5 microm in diameter) with terminal globular structures (10 microm in diameter). The hyphae were similar in width to that of an Aspergillus species but had a more moniliform appearance. Blood cultures grew a pure culture of P. obovatum. He was treated with amphotericin B and itraconazole for 6 months without remission of the diarrhea. Biopsies of the stomach, colon, and rectum showed granulomatous inflammation with marked crypt distortion simulating Crohn disease. In retrospect, the fungus was found to be resistant to both of the aforementioned drugs and susceptible to voriconazole and posaconazole. The gastrointestinal findings raise the possibility of further dissemination of a partially treated Phialemonium infection.     

Inguinal abscess caused by Rhizopus rhizopodiformis: successful treatment with surgery and amphotericin B

Rhizopus rhizopodiformis has seldom been isolated from human mucormycosis. We report the first subcutaneous abscess to be caused by this fungus. It occurred in a diabetic man and presented as an inguinal mass, suggestive of a hernia, superficial to his cadaveric renal transplant. The fungus was readily isolated from pus inoculated onto blood and chocolate agars after a short incubation. The patient was cured by surgical drainage and treatment with 2.0 g of intravenous amphotericin B. Complete identification of such isolates is recommended.     

Subcutaneous zygomycosis caused by Basidiobolus ranarum - a case report

Subcutaneous zygomycosis caused by Basidiobolus ranarum is endemic in South India. The present report is of an agricultural labourer who presented with a painless swelling of the left thigh. Culture of the affected tissue yielded B. ranarum. The swelling completely subsided with oral potassium iodide.     

Mucormycosis. Report of 3 cases

Mucormycosis is an acute and often fatal infection caused by a fungus of the Mucorales order of the Zygomycetes class. There are various clinical types, usually associated with an underlying disorder. The rhinocerebral mucormycosis is usually seen in diabetics, especially in ketoacidosis, but may also appear in healthy people. We report three cases of mucormycosis diagnosed since april 1987 through january 2001 at the Dr. Domingo Luciani Hospital, Caracas, Venezuela. Two of them had diabetes and one was apparently healthy. The most common clinical presentation of mucormycosis was the rhinocerebral infection, seen in the two diabetic patients, both of them had cavernous sinus thrombosis one with involvement of the carotid artery. The other patient with sinus involvement had no predisposing factors. All patients were treated with amphotericin B and two of them had surgical debridement of involved tissue. We emphasize the importance of an early clinical diagnosis and treatment with surgical debridement of infected tissue combined with amphotericin B.     

Disseminated Medicopsis romeroi infection in a kidney transplant recipient: A case report

Medicopsis romeroi is a rare, dematiaceous fungus that is difficult to identify using conventional fungal tests. Although uncommon, immunocompromised patients are particularly susceptible to this opportunistic fungus. Here, we report the case of a renal transplant recipient who presented with painful disseminated subcutaneous and soft tissue lesions. Sequencing of the Internal transcribed spacer (ITS) region of the ribosomal DNA identified the fungus as Medicopsis romeroi. Additionally, tissue samples from a non-healing wound on the left forearm grew Rhizopus spp. on Sabouraud dextrose agar, indicating a Mucormycosis superinfection. The patient's condition improved with surgical intervention and antifungal therapy with Posaconazole and Terbinafine. This case demonstrates the need for a high index of suspicion in order to facilitate early diagnosis and treatment and thus reduce the risk of dissemination.     

Allergic fungal rhinosinusitis caused by Neoscytalidium dimidiatum: A case report: Allergic fungal rhinosinusitis due to Neoscytalidium dimidiatum

Neoscytalidium dimidiatum is a rare dematiaceous fungus that was first described in 1916 as Dothiorella mangiferae. From the standpoint of epidemiology and therapy, early detection of fungal rhinosinusitis (FRS), the causative agents, and their associated risk factors can improve the therapeutic outcome and decrease the mortality rates among patients. In this study, we report a 34-year-old Iranian female patient with allergic bronchopulmonary aspergillosis (ABPA), who presented to our facility with an 8-year history of chronic fungal sinusitis, drug-resistant asthma, pneumonia, bronchitis, post-nasal discharge, nasal obstruction, nasal polyposis, and anemia. The patient was subjected to diagnostic nasal endoscopy and computed tomography (CT) scan of paranasal sinuses, as well as routine, complementary mycological, and molecular methods, which confirmed the diagnosis of allergic fungal rhinosinusitis in patients with ABPA. Neoscytalidium dimidiatum was isolated from the sinus of the patient. Results of in vitro susceptibility tests indicated that the case isolate was susceptible to amphotericin B and itraconazole at concentrations which are commonly achieved in patients receiving recommended dosages for invasive mycoses (0.25 to 0.75 mg/kg of body weight daily for amphotericin B and 100 to 400 mg daily for itraconazole) and resistant in vitro to caspofungin, voriconazole, and posaconazole. The patient was successfully treated with amphotericin B / itraconazole + postoperative oral corticosteroids (OCS). Neoscytalidium dimidiatum infection should be considered as a possible additional factor in the etiology of AFRS, especially in immunocompromised patients.     

Invasive Nattrassia mangiferae infections: case report, literature review, and therapeutic and taxonomic appraisal.

We report on a case of subcutaneous infection of the arm caused by the coelomycetous fungus Nattrassia mangiferae (formerly Hendersonula toruloidea) in a steroid-dependent diabetic man with chronic obstructive lung disease. The man was a resident of Arizona, where the fungus is known to be endemic on Eucalyptus camaldulensis and on citrus trees. Diagnosis of fungal infection was made by observation of narrow hyphal filaments by histopathology of biopsy specimens and isolation of a fast-growing black mold which demonstrated hyphae and arthroconidia of varying widths typical of the Scytalidium synanamorph (S. dimidiatum). The formation of pycnidia, which at maturity expressed conidia with a central median dark band, allowed for the confirmation of the isolate as N. mangiferae. Remission of the lesions occurred following intravenous therapy with amphotericin B, followed by topical clotrimazole treatment. We use this patient's case report as an opportunity to review the literature on cases of deep infection caused by Scytalidium species, to evaluate the antifungal susceptibilities of a spectrum of Scytalidium isolates, and to review the taxonomy of Scytalidium species isolated from human infections.     

Human phagocytic cell responses to Scedosporium apiospermum (Pseudallescheria boydii): variable susceptibility to oxidative injury

Scedosporium apiospermum (Pseudallescheria boydii) is an emerging opportunistic filamentous fungus that causes serious infections in both immunocompetent and immunocompromised patients. To gain insight into the immunopathogenesis of infections due to S. apiospermum, the antifungal activities of human polymorphonuclear leukocytes (PMNs), mononuclear leukocytes (MNCs), and monocyte-derived macrophages (MDMs) against two clinical isolates of S. apiospermum were evaluated. Isolate SA54A was amphotericin B resistant and was the cause of a fatal disseminated infection. Isolate SA1216 (cultured from a successfully treated localized subcutaneous infection) was susceptible to amphotericin B. MDMs exhibited similar phagocytic activities against conidia of both isolates. However, PMNs and MNCs responded differently to the hyphae of these two isolates. Serum opsonization of hyphae resulted in a higher level of superoxide anion (O(2)(-)) release by PMNs in response to SA54A (amphotericin B resistant) than that seen in response to SA1216 (amphotericin B susceptible; P < 0.001). Despite this increased O(2)(-) production, PMNs and MNCs induced less hyphal damage to SA54A than to SA1216 (P < 0.001). To investigate the potential mechanisms responsible for these differences, hyphal damage was evaluated in the presence of antifungal oxidative metabolites as well as in the presence of a series of inhibitors and scavengers of antifungal PMN function. Mannose, catalase, superoxide dismutase, dimethyl sulfoxide, and heparin had no effect on PMN-induced hyphal damage to either of the two isolates. However, azide, which inhibits PMN myeloperoxidase activity, significantly reduced hyphal damage to SA1216 (P < 0.01) but not to SA54A. Hyphae of SA1216 were slightly more susceptible to oxidative pathway products, particularly HOCl, than those of SA54A. Thus, S. apiospermum is susceptible to antifungal phagocytic function to various degrees. The selective inhibitory pattern of azide with respect to hyphal damage and the parallel susceptibility to HOCl suggests an important difference in susceptibilities to myeloperoxidase products that may be related to the various levels of pathogenicity and amphotericin B resistance of S. apiospermum.     

Pulmonary colonization by Chrysosporium zonatum associated with allergic inflammation in an immunocompetent subject

We report a case of noninvasive pulmonary disease due to Chrysosporium zonatum in an immunocompetent male. The fungus colonized an existing tuberculous cavity and was isolated from transbronchial lavage fluid and from a percutaneous aspiration specimen. The disease was accompanied by the unusual feature of an allergic reaction. The fungus ball was successfully treated by intracavitary administration of amphotericin B. C. zonatum is the anamorph of the heterothallic ascomycete Uncinocarpus orissi, and the identity of the case isolate was verified by formation of ascospores in mating tests with reference isolates.     

Fulminating fungal sinusitis caused by Valsa sordida, a plant pathogen, in a patient immunocompromised by acute myeloid leukemia.

We describe a case in which a patient immunocompromised by acute myeloid leukemia experienced acute fulminating invasive fungal sinusitis followed by pneumonia and then death. Though the microbiology of the pneumonia could not be directly investigated, nasal lesions revealed fungal mycelium. Valsa sordida was consistently cultured from a biopsied sample. The fungus was identified to the genus level based on morphology in culture and DNA sequence homology, and then was placed at species level by means of phylogenetic analysis of the nuclear ribosomal internal transcribed spacer region. The fungus is in the order Diaporthales, family Valsaceae in the Ascomycota and is distributed worldwide as a pathogen of trees in the genera Populus and Salix. Koch's postulates were demonstrated to apply in a neutropenic rat model. The fungus was susceptible to antifungals with MIC-0 scores of 0.0313 microg/ml for amphotericin B, 0.25 microg/ml for voriconazole, 0.0313 microg/ml for caspofungin, and MIC-2 of 16 microg/ml for fluconazole. This is the first substantiated report of an isolate in the genus Valsa (anamorph Cytospora) being identified in human disease.     

Basidiobolomycose probable chez un jeune rural togolais traitée avec succès par du kétoconazole

Basidiobolomycosis is a deep mycosis which preferentially affects rural young people in tropical countries. We report a case of basidiobolomycosis successfully treated with ketoconazole. It was a 9-year-old boy of rural origin in whom the diagnosis of basidiobolomycosis was suspected due to a deep skin infiltration involving the chest and neck. Histology revealed hypodermic granulomatous inflammation with predominantly macrophage and eosinophils. The child was treated successfully with ketoconazole in eight weeks. Treatment of basidiobolomycosis is based on azole derivatives which are particularly effective. Histopathology is very important in the diagnosis of this affection, especially in tropical countries where it may simulate Mycobacterium ulcerans infection.     

Severe pigmented keratitis caused by Cladorrhinum bulbillosum

We report a case of severe pigmented keratitis with poor prognosis, caused by Cladorrhinum bulbillosum. Antifungal treatment with topical natamycin and fluconazole eye drops and oral tablet fluconazole failed to heal the ulcer and resulted in perforation. The causative fungus, C. bulbillosum, was identified on the basis of its typical microscopic features and 98% sequence homology to ex-type isolate CBS 304.90 (accession no. FM955448). The results of an in vitro antifungal susceptibility test indicated that the isolate was susceptible to natamycin, amphotericin B, fluconazole and itraconazole. The present case is the third case of keratitis and the second case of human keratitis. Compromised immunity due to liver cirrhosis could lead to a failed prognosis even when the fungal isolate is highly susceptible to antifungal treatment.     

An unusual localization of mucormycosis diagnosed by fine needle aspiration cytology: a case report

Mucormycosis is an uncommon fungal infection, occurring mainly in patients with acidosis, chronic illnesses and malignancies. The most frequent site of involvement in patients of hematological malignancies is the respiratory tract. Isolated subcutaneous localization of mucormycosis in such patients is extremely rare. We report a case of a young patient of non-Hodgkin's lymphoma on chemotherapy who presented with a subcutaneous swelling on the anterior aspect of right thigh. Fine needle aspiration cytology (FNAC) smears from the swelling revealed numerous characteristic broad, irregularly contoured and pleomorphic hyphae of mucormycosis. This fungus seldom grows in culture and confirmation of the diagnosis depends on cytological or histological examination of infected tissues. Our case report documents a rare site of isolated mucormycosis infection and emphasizes the role of FNAC as a simple, rapid, accurate, and useful method of diagnosing fungal infections.