Navigating treatment for basidiobolomycosis: a qualitative review of 24 cases.

BACKGROUND AND OBJECTIVES: Zygomycosis, a severe form of fungal infection, is classified into two categories: Mucorales and Entomophthorales. Within the Entomophthorales category, Basidiobolomycosis is a rarely recognized genus that can have significant health implications. Prompt diagnosis and appropriate treatment, which includes the use of antifungal medication and surgical procedures, are vital for enhancing the prognosis of patients. The objective of this study is to investigate the response to treatment in patients hospitalized due to basidiobolomycosis. METHODS: We carried out a retrospective study, in which we analyzed data from 49 patients who were diagnosed with Entomophthorale, Zygomycosis, and Basidiobolomycosis at Namazi Hospital, Shiraz, between the years 1997 and 2019. The data included parameters such as demographic information, clinical symptoms, imaging findings, treatment methods, and patient outcomes. RESULTS: Out of 49 patients, 24 children, predominantly male (83.3%), were definitively diagnosed with basidiobolomycosis. The ages of the patients ranged from 1 to 16 years, with an average of 5.75 years. The most frequently observed clinical manifestations included abdominal pain (70.8%), fever (54.2%), hematochezia (41.7%), vomiting (20.8%), and anorexia (16.7%). Half of the patients exhibited failure to thrive (FTT), while abdominal distension was present in 25% of the cases, and a palpable abdominal mass was found in 37% of the patients. The primary treatment strategy incorporated surgical interventions complemented by a comprehensive antifungal regimen. This regimen included medications such as amphotericin B, cotrimoxazole, itraconazole, potassium iodide, and voriconazole. These were mainly administered in a combination therapy pattern or as a monotherapy of amphotericin B. Twenty-two patients were discharged, while two patients died due to complications from the disease. CONCLUSION: Our findings indicate that the prevailing treatment modalities generally involve surgical intervention supplemented by antifungal regimens, including Amphotericin B, Cotrimoxazole, Potassium Iodide, and Itraconazole.

Disseminated Basidiobolomycosis Caused by Basidiobolus omanensis in a Child with Acute Lymphoblastic Leukemia (ALL). Case Report and Literature Review.

Basidiobolomycosis is an uncommon fungal infection caused by the genus Basidiobolus. In immunocompetent children, it usually causes cutaneous infection and rarely affects the gastrointestinal tract, and it is extremely rare for the disease to spread. The present study reports the first case of disseminated basidiobolomycosis caused by Basidiobolus omanensis in a child with acute lymphoblastic leukemia who died as a result of uncontrolled infection and multi-organ failure despite surgical and antifungal therapy with L-AMB and voriconazole. A review of the literature yielded 76 cases, including the current case with the majority of which were reported as invasive gastrointestinal infection. The median age was 4 years (61 male and 15 female) and the majority of these children were from the Middle East (80%), specifically Saudi Arabia (45%). Most patients were treated with systemic antifungal agents (mostly itraconazole and amphotericin B). Surgical intervention was done in 25% of these patients and the death rate was 12%.

The gastrointestinal basidiobolomycosis, a rare entity: Case report and review of literature.

Gastrointestinal basisdiobolomycosis is a rare fungal infection prevalent in the tropical and subtropical regions. It has variable clinical manifestations that present a challenge for timely diagnosis. Herein, we present a case of gastrointestinal basidiobolomycosis in a 34-year-old male. To the best of our knowledge, it is the first reported case of gastrointestinal basidiobolomycosis from Pakistan. The patient presented with abdominal pain and was first operated for perforated appendix and then for a mesenteric mass based on the findings of CT scan. Histopathologic examination showed broad septate fungal hyphae surrounded by eosinophilic proteinaceous material (Splendore-Hoppeli phenomenon), neutrophils, and histiocytes. Diagnosis of gastrointestinal basidiobolomycosis was established on the basis of this morphology.

Pediatric case of conidiobolomycosis: A rare entity.

Conidiobolomycosis caused by Conidiobolus species is an uncommon infection restricted to tropical and subtropical regions, usually affecting immunocompetent individuals. More than half of pediatric cases of conidiobolomycosis across the globe are from India. We report a case of subcutaneous conidiobolomycosis in an adolescent with development delay who responded to combined therapy with itraconazole and saturated solution of potassium iodide.

Rhinofacial conidiobolomycosis: Clinical and microbiological characterisation and shift in the management of a rare disease.

BACKGROUND: Conidiobolomycosis is a rare tropical rhinofacial fungal infection which has not been well characterised. The available evidence in its management is sparse due to lack of clinical studies and the limited data on antifungal susceptibility patterns. OBJECTIVE: To analyse the clinical manifestations, antifungal treatment and outcomes of patients with conidiobolomycosis and to determine antifungal susceptibility profiles of the isolates. PATIENTS/METHODS: Retrospective analysis of data of all patients with a diagnosis of conidiobolomycosis confirmed by histopathology and culture at a tertiary care hospital from 2012 to 2019 was done. RESULTS: There were 22 patients, 21 males and one female, with a mean age of 37.1 years. Most common presenting symptom was nasal obstruction, found in 20 (90.90%) patients. Patients who presented within 12 months had a better cure rate (85%) compared to those who presented late (67%). Among the 19 patients who had a follow-up, good outcome was seen in 15 of the 17 (88.24%) patients who were on itraconazole or potassium iodide containing regimen. Of the six patients who received additional trimethoprim-sulphamethoxazole (co-trimoxazole), 67% showed good outcome with two patients showing complete cure and two patients still on treatment with significant improvement. High minimum inhibitory concentration (MIC) values were noted for azoles and amphotericin B, whereas co-trimoxazole showed lowest MIC ranges. CONCLUSION: Itraconazole and potassium iodide are reasonable first-line options for the treatment of conidiobolomycosis. Good clinical response to KI and comparatively lower MIC of co-trimoxazole are promising. Further studies are required for developing clinical breakpoints that can predict therapeutic outcomes.

An Infant with Acute Bloody Diarrhea and Gastrointestinal Basidiobolomycosis: An Unusual Presentation of a Rare Disease.

Basidiobolomycosis is a fungal infection caused mainly by Basidiobolus ranarum, a filamentous fungus of the order Entomophthorales and the family Basidiobolaceae. This infection typically involves the skin and soft tissue; however, visceral organ involvement has also been reported. Here, we report a case of gastrointestinal basidiobolomycosis in a young child who presented with acute bloody diarrhea which was initially misdiagnosed as intussusception.

Conidiobolus pachyzygosporus invasive pulmonary infection in a patient with acute myeloid leukemia: case report and review of the literature.

BACKGROUND: Conidiobolus spp. (mainly C. coronatus) are the causal agents of rhino-facial conidiobolomycosis, a limited soft tissue infection, which is essentially observed in immunocompetent individuals from tropical areas. Rare cases of invasive conidiobolomycosis due to C. coronatus or other species (C.incongruus, C.lamprauges) have been reported in immunocompromised patients. We report here the first case of invasive pulmonary fungal infection due to Conidiobolus pachyzygosporus in a Swiss patient with onco-haematologic malignancy. CASE PRESENTATION: A 71 year-old female was admitted in a Swiss hospital for induction chemotherapy of acute myeloid leukemia. A chest CT performed during the neutropenic phase identified three well-circumscribed lung lesions consistent with invasive fungal infection, along with a positive 1,3-beta-d-glucan assay in serum. A transbronchial biopsy of the lung lesions revealed large occasionally septate hyphae. A Conidiobolus spp. was detected by direct 18S rDNA in the tissue biopsy and subsequently identified at species level as C. pachyzygosporus by 28S rDNA sequencing. The infection was cured after isavuconazole therapy, recovery of the immune system and surgical resection of lung lesions. CONCLUSIONS: This is the first description of C. pachyzygosporus as human pathogen and second case report of invasive conidiobolomycosis from a European country.

Gastrointestinal basidiobolomycosis in a patient suffering from duodenal ulcer with perforation: First case report from Italy.

Gastrointestinal basidiobolomycosis (GIB), an unusual fungal infection caused by the fungus Basidiobolus ranarum, is rarely reported in the medical literature. GIB is difficult to diagnose because its clinical presentation is non-specific and has no identifiable risk factors. We report here the first case of GIB diagnosed in Italy in a patient suffering from a duodenal ulcer with perforation. The patient was successfully treated with itraconazole. The absence of non-specific signs and symptoms of GIB may delay a definitive diagnosis and treatment. A microbiological investigation should always be requested in order to reach a rapid and definitive diagnosis and to rule out other intestinal diseases.

Experimental infection in gerbils by Conidiobolus lamprauges.

Conidiobolomycosis is an emerging entomophthoramycosis caused by fungi Conidiobolus spp. Animal models are essential for the study of infectious disease in various areas such as pathogenesis, diagnostic methods, treatment and prevention. There is not currently an animal model for conidiobolomycosis. The aim of this study was to create an experimental infection protocol for Conidiobolus lamprauges in gerbils (Meriones unguiculatus). The study animals were randomly divided into four groups of four animals: immunosuppressed with cyclophosphamide (CPA) and infected with C. lamprauges (G1), immunocompetent and infected with C. lamprauges (G2), immunosuppressed with CPA (G3), and an immunocompetent control group (G4). Clinical signs were observed only in G1 animals, where the mortality rate reached 75% by day 7 after infection (AI) with a median survival of 2 days. C. lamprauges was detected only in G1, both by PCR and by isolation. Necropsies of the G1 animals showed lesions in the nasal cavity and lung tissue. These lesions were characterized by polymorphonuclear infiltrate cells and by the presence of hyphal structures under silver staining. This animal model will be useful for further investigation of diseases caused by C. lamprauges, particularly of those associated with immunosuppression factors in naturally occurring animal infections.

Pathogenicity of Conidiobolus coronatus and Fusarium solani in mouse models.

To study the pathogenicity of Conidiobolus coronatus (C. coronatus) and Fusarium solani (F. solani) in animal models. Immunocompromised mice were treated with cyclophosphamide and prednisolone via intraperitoneal injection before and after inoculation. According to pathogenic characteristics of different fungi, C. coronatus was used to infect mice via intravenous inoculation, intraperitoneal inoculation, gastrointestinal infusion and intradermal inoculation methods. And F. solani was used to infect mice by inoculation via the abraded or normal skin. In the group of immunocompromised mice, C. coronatus was isolated from the lung tissues of one mouse on day 7 and another on day 10 respectively. The corresponding histopathology revealed infiltration of local inflammatory cells in the lung tissue. Pathogenic lesions were observed in all normal and immunocompromised mice infected with F. solani via abraded skin. The lesions in the immunocompromised mice were more severe and persisted longer than those in the normal mice. Moreover, hyphae were mostly observed in the histopathological examination and fungal culture from the immunocompromised mouse. The pathogenicity of C. coronatus was relatively weak as it did not induce local infections and did not disseminate the disease in immunocompetent and immunocompromised mice. Therefore, F. solani is a type of opportunistic pathogenic fungus, and abraded skin is one of the causative routes of infection.

Subcutaneous Basidiobolomycosis Resembling Fournier's Gangrene.

Basidiobolomycosis is an uncommon cutaneous zygomycete infection typically seen in immunocompetent individuals. Diagnosis can be made by biopsy and fungal culture of the lesion. Treatment with Potassium iodide and co-trimoxazole is simple and effective. Early and accurate diagnosis of basidiobolomycosis is essential to avoid dissemination and mortality. We present a case with basidiobolomycosis resembling Fournier's gangrene.

Abdominal imaging findings in gastrointestinal basidiobolomycosis.

PURPOSE: To describe the abdominal imaging findings of patients with gastrointestinal Basidiobolus ranarum infection. METHODS: A literature search was performed to compile the abdominal imaging findings of all reported worldwide cases of gastrointestinal basidiobolomycosis (GIB). In addition, a retrospective review at our institution was performed to identify GIB cases that had imaging findings. A radiologist aware of the diagnosis reviewed the imaging findings in detail. Additional information was obtained from the medical records. RESULTS: A total of 73 GIB cases have been published in the medical literature. The most common abdominal imaging findings were masses in the colon, the liver, or multiple sites and bowel wall thickening. Initially, many patients were considered to have either a neoplasm or Crohn disease. We identified 7 proven cases of GIB at our institution, of which 4 had imaging studies (4 computed tomography [CT] examinations, 4 abdominal radiographs, and an upper gastrointestinal study). Imaging studies showed abnormalities in all 4 cases. Three-fourths of our study patients had an abdominal mass at CT. Two of 3 masses involved the kidneys and included urinary obstruction. All masses showed an inflammatory component with adjacent soft tissue stranding, with or without abscess formation. CONCLUSIONS: Radiologists should consider GIB when a patient from an arid climate presents with abdominal pain, weight loss, and an inflammatory abdominal mass on CT. Abdominal masses of the colon or liver, bowel wall thickening, and abscesses are the most common imaging findings.

Effects of 7-hydroxycalamenene isolated from Croton cajucara essential oil on growth, lipid content and ultrastructural aspects of Rhizopus oryzae.

The leaves and bark of Croton cajucara, a shrub from the Amazon region, have been used in folk medicine to treat diabetes, malaria, and gastrointestinal and liver disorders. The essential oil from the leaves, rich in linalool, presented antileishmanial and antimicrobial activities. A chemotype of this species was found with an essential oil rich in 7-hydroxycalamenene. During our studies of the C. cajucara essential oil, we isolated 7-hydroxycalamenene at > 98 % purity. The minimum inhibitory concentration of 7-hydroxycalamenene against Absidia cylindrospora, Cunninghamella elegans, Mucor circinelloides, Mucor circinelloides f. circinelloides, Mucor mucedo, Mucor plumbeus, Mucor ramosissimus, Rhizopus microsporus, Rhizopus oryzae, and Syncephalastrum racemosum ranged from 19.53 to 2500 µg/mL. The reference drug used, amphotericin B, presented a minimum inhibitory concentration ranging from 0.085 µg/mL to 43.87 µg/mL. 7-Hydroxycalamenene also altered spore differentiation and total lipid content. Ultrastructural analysis by transmission electron microscopy showed significant alterations in the cellular structure of R. oryzae.

Entomophthoromycosis: a challenging emerging disease.

Entomophthoromycosis is a rare fungal infection that may affect immunocompetent hosts; predominantly in tropical and subtropical regions. Recently, the importance of this emerging mycosis has increased and the scope of its manifestations has been expanded. These manifestations; however, may masquerade as other clinical entities. Prompt diagnosis of this infection requires a high index of suspicion. Although histopathological examination and cultures are the gold standard diagnostic tools; molecular diagnosis is now available and started to play an important role. The cornerstone treatment is prolonged anti-fungal therapy along with surgical debridement. More awareness of this mycosis is warranted for definitive diagnosis and implementation of early proper therapeutic strategies.

Diagnosis of gastrointestinal basidiobolomycosis: a mini-review.

Basidiobolus ranarum (Entomophthoromycotina) very rarely affects the gastrointestinal (GI) tract. To date, reported paediatric GI basidiobolomycosis cases are 27 worldwide; 19 from Saudi Arabia and 8 from other parts of the world. Often these cases present a diagnostic dilemma, are prone to misdiagnosis and lack of disease confirmation by proper molecular methodologies. The fungal mass removed by surgery is usually sent for conciliar histopathology, isolation by fungal cultures and final molecular testing for basidiobolomycosis. The incidence of basidiobolomycoses, their predisposing factors and the molecular diagnosis of the fungus causing the disease in combination with a phylogenetic framework are reviewed.

Cutaneous fungal infection in a neonatal intensive care unit patient: a case report and literature review.

Fungal skin infections are not uncommon in healthy, premature or immunocompromised newborns. Healthy neonates usually develop fungal skin infections caused by dermatophytes, Candida and Malassezia species, whereas immunocompromised neonates are more susceptible to skin infections with opportunistic pathogens (Aspergilus, Zygomycetes). Therefore neonatal fungal skin infections can range from generally benign superficial lesions to potentially fatal, deep, necrotic forms with dissemination. We present the case of a premature neonate twin with cutaneous fungal infection in a neonatal intensive care unit. Because there were doubts concerning the correspondence of the clinical features with the cultured species in the newborn, a literature review was performed searching for similar clinical cases.

Successful treatment of gastrointestinal basidiobolomycosis with voriconazole without surgical intervention.

Basidiobolomycosis is a rare disease due to fungus Basidiobolus ranarum, an environmental saprophyte that is found worldwide, though mainly reported in the tropical and subtropical regions. Basidiobolomycosis is an unusual fungal skin infection, rarely involves the gastrointestinal (GI) tract. Most of the cases of paediatric GI basidiobolomycosis (GIB) were reported from the southern region of Saudi Arabia. We report an 11-year-old Saudi boy. He presented with a huge right lower quadrant abdominal mass and marked eosinophilia. Abdominal computed tomography scan revealed a large caecal mass. A biopsy was taken and it showed transmural granulomatous inflammation. A diagnosis of GIB was confirmed by specific features in histopathology. Most of the reported paediatric cases with GIB required adjuvant therapy of antifungal and surgical resection. In our case, treatment with voriconazole alone for 1 year was successful with complete recovery and with no recurrence after a year of discontinuing the treatment.

Subcutaneous zygomycosis in children: 2 case reports.

Basidiobolus ranarum is an uncommon subcutaneous zygomycosis mostly found in immunocompetent children in tropical countries. Presence of slow growing non-tender, non-inflammatory, subcutaneous swelling that does not spread beyond the subcutaneous tissue are classic clinical features. The authors report two cases of subcutaneous zygomycosis which tissue cultures were positive for Basidiobolus ranarum. The first case was a 10-months-old boy presented with prolonged high fever and a rapidly expanding ulcerated plaque unresponsive to systemic antibiotic. The second case was a 2-years-old girl presented with slow expanding mass at the buttock. Histopathology of both cases showed lobular panniculitis with eosinophilic infiltration and fungal culture revealed Basidiobolus ranarum. Oral itraconazole was given with good clinical response in both cases.