Subcutaneous zygomycosis in an immunocompetent patient caused by Basidiobolus ranarum.

Zygomycosis is a fungal infection in humans caused by orders Mucorales and Entomophthorales. The incidence of Mucorales causing mucormycosis is on a rise and is well documented, whereas Entomophthorales is rare. Among Entomophthorales, infections caused by Conidiobolus are more common than Basidiobolus Here we present a case of subcutaneous basidiobolomycosis in a female patient. The patient had hyperpigmentation in the thigh region for 6 months and serous discharge for 4 months. All initial findings suggested the inflammatory stage of morphea. Differential diagnoses of granuloma annulare, malignant melanoma and morphea were considered radiologically. A good suspicion of fungal aetiology by the dermatologist led to an appropriate diagnosis of subcutaneous basidiobolomycosis based on fungal culture and histopathological examination. Based on macroscopic and microscopic findings, the causative organism was confirmed to be Basidiobolus ranarum The patient was started on oral potassium iodide and itraconazole and showed a good prognosis.

Basidiobolus haptosporus-like fungus as a causal agent of gastrointestinal basidiobolomycosis.

Basidiobolus species were isolated from colonic biopsy samples of patients with gastrointestinal basidiobolomycosis (GIB) in southern Saudi Arabia. Isolated fungi were initially identified using classical mycological tools and confirmed by sequence analysis of the large subunit ribosomal RNA gene. Phenotypic tests revealed zygomycete-like fungi which conform to those of Basidiobolus species. Five sequenced strains formed a monophyletic clade in the 28S ribosomal RNA gene phylogenetic tree. They shared 99.97% similarity with B. haptosporus and 99.97% with B. haptosporus var. minor, and relatively lower similarity with B. ranarum (99.925%). The study suggests a new and a serious causal agent of GIB related to Basidiobolus haptosporus. These isolates are not related to B. ranarum, which is commonly linked to this disease.

Antifungals in a case of basidiobolomycosis: role and limitations.

A 10-year-old Saudi boy was diagnosed to have basidiobolomycosis after a stormy course of his ailment. Therapy was initiated with intravenous antifungal, voriconazole, which was well tolerated for 6 weeks except for local excoriation at the site of ileostomy. He developed drug-induced hepatitis on oral voriconazole, therefore, switched to oral itraconazole following which he experienced severe chest pain. Alternatively, co-trimoxazole (bactrim) an antibacterial with antifungal activity was prescribed but he had the intolerance to it as well. Unfortunately, posaconazole as an alternative antifungal was not available in our centre. We report here a Saudi boy who developed an intolerance to most common antifungals used clinically 6 weeks after the therapy was initiated.

Gastrointestinal basidiobolomycosis: An emerging mycosis difficult to diagnose but curable. Case report and review of the literature.

BACKGROUND: Gastrointestinal basidiobolomycosis (GIB) is a rare mycosis affecting almost exclusively immunocompetent subjects. METHODS: We describe a case of GIB caused by Basidiobolus ranarum in a 25-year-old Italian immunocompetent man resident in Ireland who presented a 2-month history of epigastric pain. Suspecting colon cancer he underwent a right hemicolectomy subsequently leading to a diagnosis of GIB by means of molecular biology. After surgery a 9-month therapy with itraconazole was employed with a good outcome. A review of medical literature regarding GIB cases published in the period 1964-2017 is presented. RESULTS: One-hundred and two cases of GIB were included in this analysis. The disease was observed predominantly in male gender (74.5%) and children (41.2%). Abdominal pain was the single most common complaint (86.3%) followed by fever (40.2%) and evidence of an abdominal mass (30.4%). Peripheral blood eosinophilia was detected in 85.7% of cases. Most of the patients were diagnosed in Saudi Arabia (37.2%) followed by USA (21.6%) and Iran (20.6%). Surgery plus antifungal therapy was employed in the majority of patients (77.5%). An unfavourable outcome was documented globally in 18.6% of patients. CONCLUSIONS: GIB seems to be an emerging intestinal mycosis among immunocompetent patients living in the Middle East and Arizona.

Gastro-intestinal basidiobolomycosis in a 2-year-old boy: dramatic response to potassium iodide.

Gastro-intestinal basidiobolomycosis (GIB) is a rare fungal infection caused by Basidiobolus ranarum. Treatment includes surgical resection and long-term antifungal therapy. A 2.5-year-old boy presented with a 10-day history of abdominal pain, fever and diarrhoea, and a palpable abdominal mass was detected. Resection was undertaken and histology confirmed basidiobolomycosis. Treatment with amphotericin B and itraconazole was commenced, but the infection progressed and spread to involve the intestines, liver, ribs and lung, and also the abdominal wall after 6 months, requiring four operative procedures. Because of unresponsiveness to amphotericin and itraconazole, oral potassium iodide was added which resulted in complete resolution of the infection. Potassium iodide is an essential component of the treatment of systemic B. ranarum.

Basidiobolomycosis complicated by hydronephrosis and a perinephric abscess presenting as a hypertensive emergency in a 7-year-old boy.

A 7-year-old boy presented with a chronic, indurated, tender left thigh swelling in association with a hypertensive emergency. He had a bilateral moderate degree of hydronephrosis and a left perinephric abscess, and MRI features of posterior reversible encephalopathy syndrome. Histopathological examination of the biopsy specimen demonstrated eosinophilic fasciitis with filamentous fungi. Basidiobolus ranarum was isolated from the culture. The fungus was also isolated from a perinephric fluid aspirate. Computerised tomography of the abdomen demonstrated features consistent with fungal invasion of the pelvic floor muscles and urinary bladder, leading to bilateral hydronephrosis. He required multiple antihypertensive drug therapy and was treated with intravenous amphotericin B, oral itraconazole and potassium iodide. Antihypertensive agents were discontinued after 2 weeks of antifungal therapy. At 6-months follow-up, the hydronephrosis had resolved completely. Perinephric abscess associated with basidiobolomycosis has not been reported previously.

Cutaneous basidiobolomycosis: Seven cases in southern Benin.

BACKGROUND: Cutaneous basidiobolomycosis is the most common form of entomophthoramycosis. Herein we report seven cases of cutaneous basidiobolomycosis. PATIENTS AND METHODS: A retrospective observational study was conducted at the Buruli ulcer treatment centre in Pobè and at the national teaching hospital in Cotonou from 2010 to 2015. RESULTS: Seven cases of cutaneous basidiobolomycosis were diagnosed. The mean patient age was 9.53 years. There were 4 female and 3 male patients, all from southeast Benin. Clinically, the disease presented in all cases as a hard, well-defined, subcutaneous plaque with little inflammation, and which could easily be lifted from the deep structures but remained attached to the surface structures. The overlying skin was hyperpigmented. Plaques were localized to the buttocks or thighs. All patients had inflammatory anaemia with an accelerated erythrocyte sedimentation rate (30 to 70mm over the first hour), and a low haemoglobin count (8.7 to 11.4g/dL). Blood hypereosinophilia (650 to 3784elements/mm3) was present in six of the seven subjects. Histopathology (performed for 5 of the 7 subjects) showed granulomatous lesions with foreign-body giant cells, and inflammatory cells, with occasional eosinophils surrounding fungal hyphae (Splendore-Hoeppli phenomenon). Mycological analysis revealed Basidiobolus ranarum in three cases. The patients were treated with ketoconazole (5/7) and itraconazole (2/7), with good outcomes after 10 to 24 weeks of therapy. DISCUSSION: Cutaneous basidiobolomycosis is uncommon in southern Benin, with only seven cases being diagnosed over 6 years. The diagnosis of cutaneous basidiobolomycosis is a challenge in the field in Benin due to the non-specific clinical presentation, the lack of technical resources, and the existence of numerous differential diagnoses. CONCLUSION: Cutaneous basidiobolomycosis is an uncommon fungal infection in southern Benin chiefly affecting children.

[A case of rhinofacial entomophthoromycosis in Soudano-Sahelian tropical climate in Burkina Faso]. / Un cas d'entomophthoromycose rhinofaciale en climat tropical soudano-sahélien au Burkina Faso.

We describe a rhinofacial entomophthoramycosis case in a sexagenarian (65 years old) housewife. She was immunocompetent and resident of Burkina Faso. She consulted both the service of dermatology and the service of stomatology of the Teaching Hospital of Bobo-Dioulasso in February 2016 for a diffuse facial tumefaction evolving over six months. This tumefaction was associated with headaches and a left nasal obstruction. Histological examination of the lesion showed an important and polymorphic inflammatory reaction. Also, a filamentous fungus with wide non-septated hyphae and right-angled fungal branching, consistent with mucormycosis was isolated. Mycological diagnosis based on fungal culture with Sabouraud medium without any antibiotic and cyclohexemide after incubation at 27°C and at 30°C was negative. Furthermore, it was not possible to amplify the DNA extracted from biopsy. Antifungal therapy based on the administration of fluconazole per os at 800mg/day was started allowing clinical improvement. This is the first case of a rhinofacial entomophtharomycosis documented in Bobo-Dioulasso. Rhinofacial entomophthoromycosis is largely unknown, even in tropical regions such as Burkina Faso. This lack of knowledge results in a delay in the diagnosis, and subsequently a bad prognosis. It is therefore urgent to improve knowledge on this disease to guide diagnostic steps, prognosis of outcome, and antifungal therapy.

Survival Without Neurological Impairment of a Patient With Rhino-Orbito-Cerebral Zygomycosis.

Rhinocerebral zygomycosis is a rare condition characterized by infection initially in the nose and paranasal sinuses with atypical symptoms consistent with sinusitis. Once established in the nasal sinuses, the infection can easily spread to the orbital region and brain. In the localized form of the infection, the mortality rate is approximately 10%, which increases in the occurrence of orbital involvement and survival in patients of cerebral dissemination is extremely rare. The present paper reports a patient of nasal zygomycosis with orbital and cerebral dissemination in an otherwise healthy patient who survived after 14 months of hospitalization with no neurological impairment. A review of the literature addressing aspects related to diagnosis, treatment, and complications of this fungal infection is also presented.

Entomophthoramycosis: a neglected tropical mycosis.

The term 'entomophthoramycosis' classically refers to infections caused by members of the order Entomophthorales. A new subphylum, Entomophthoramycota, has been created to include Basidiobolomycetes, Neozygitomycetes and Entomophthoramycetes. Basidiobolomycetes encompass Basidiobolus spp., while the Entomophthoramycetes include Conidiobolus spp. Conidiobolus spp. characteristically cause rhinofacial entomophthoramycosis in apparently immunocompetent hosts. Conidiobolus spp. may also cause disseminated infection in immunocompromised patients. Basidiobolus spp. more typically cause subcutaneous entomophthoramycosis of the limbs, buttocks, back and thorax in immunocompetent patients. While once considered to be rare, there is an increasing number of reported cases of gastrointestinal infection caused by Basidiobolus spp. worldwide in countries such as United States, Thailand, Australia, Iran, Egypt and Saudi Arabia. These cases have clinical presentations similar to those of inflammatory bowel diseases, particularly Crohn's disease. Retroperitoneal, pulmonary, nasal and disseminated basidiobolomycosis have also been reported. Histology of entomophthoramycosis may reveal the Splendore-Hoeppli phenomenon. Culture of infected tissue remains the definitive method of laboratory diagnosis. However, molecular methods with specific DNA probes and panfungal primers, as well as real time PCR, are increasingly used to detect and identify these organisms in tissue. Treatment largely consists of therapy with antifungal triazoles. Surgery plays a selective role in the management of entomophthoramycosis, depending upon location, organism and extent of the infection.

Visceral basidiobolomycosis: An overlooked infection in immunocompetent children.

Visceral basidiobolomycosis is an unusual fungal infection of viscera caused by saprophyte Basidiobolus ranarum. It is very rare in healthy children and poses a diagnostic challenge due to the non-specific clinical presentation and the absence of predisposing factors. We report a case of gastrointestinal basidiobolomycosis in a 4-year-old healthy girl who presented with a short history of abdominal pain, bleeding per rectum, fever, and weight loss. The diagnosis was based on high eosinophilic count, classical histopathology findings of fungal hyphae (the Splendore-Hoeppli phenomenon), and positive fungal culture from a tissue biopsy. Fungal infection was successfully eradicated with a combined approach of surgical resection of the infected tissue and a well-monitored course of antifungal therapy. The atypical clinical presentation, diagnostic techniques, and the role of surgery in the management of a rare and lethal fungal disease in an immunocompetent child are discussed.

Chronic rhinofacial basidiobolomycosis caused by Basidiobolus ranarum: Report of a case from Aseer Region, Kingdom of Saudi Arabia.

We present a case of fungal sinusitis caused by Basidiobolus ranarum in a 22-year-old male patient with chronic rhinosinusitis in Aseer region, Kingdom of Saudi Arabia. The patient was admitted with nasal obstruction accompanied by itching, sneezing, rhinorrhea, epistaxis and recurrent headache. Axial computed tomography (CT) scan of the paranasal sinuses showed a clear left facial swelling chronic inflammation and granulomata. Basidiobolus ranarum fungus was isolated on Sabouraud dextrose agar from a biopsy specimen. The organism was characterized by flat, yellowish-grey, glabrous, becoming radially folded fungus that under the microscope showed broad vegetative hyaline hyphae that bear zygospores with protuberances. The patient made good recovery and was discharged home with no recurrences after receiving oral itraconazole and removal of the polyps surgically.

Gastrointestinal basidiobolomycosis in a dog.

An 8-year-old, spayed, female Shiba dog was presented to a referring veterinarian with a complaint of chronic diarrhea and anorexia. Ultrasound and radiographs revealed an irregular mass in the pelvic cavity. The mass and the affected section of colon were surgically removed. Histopathological examination revealed multifocal coalescing granulomas and effaced intestinal structures. Central necrotic debris surrounded by multinucleated giant cells, lymphocytes, plasma cells and neutrophils was observed. Numerous, irregularly branched hyphae with pale basophilic, thin walls and occasional bulbous enlargements at the tips were present. Polymerase chain reaction identified Basidiobolus ranarum, successfully confirming a definitive diagnosis of basidiobolomycosis. To the best of our knowledge, this is the first report of intestinal basidiobolomycosis in a dog.

Fungal inflammatory masses masquerading as colorectal cancer: a case report.

BACKGROUND: Non malignant invasive tumors of the colon and rectum are very rare. Gastrointestinal Basidiobolomycosis can present as a mass lesion mimicking colorectal cancer. CASE PRESENTATION: A 56 year old Caucasian male was evaluated for abdominal and pelvic pain for 4 weeks complicated by acute urinary retention. Radiological evaluation showed him to have recto-sigmoid and cecal mass. Endoscopic examination and biopsies did not reveal a definite diagnosis. Computerized tomography guided biopsy of the mass showed fungal elements consistent with gastrointestinal basidiobolomycosis. He was treated with Itraconazole for 12 months with very good clinical and radiological response. CONCLUSION: Basidiobolomycosis of the gastrointestinal tract should be considered during evaluation of colorectal masses with atypical presentation. It is a rare entity seen more in endemic regions of the world for basidiobolomycosis including southwestern United States.

When you can't see the wood for the trees. Mucor circinelloides: A rare case of primary cutaneous zygomycosis.

A patient with refractory diffuse lymphoma treated for pulmonary invasive aspergillosis developed a concomitant primary cutaneous mucormycosis. The mucormycete was identified by sequencing as Mucor circinelloides. This case confirms the importance of a rapid pathogen diagnosis in immunocompromised patients and the usefulness of molecular methods for identification of rare fungal species.