Ileocecal basidiobolomycosis mimicking malignancy successfully treated without surgery: A case report

Gastrointestinal basidiobolomycosis (GIB) is an unusual fungal infection that manifests in the skin and rarely involves other systems. All of the few cases of GIB reported so far were diagnosed with difficulty, necessitating laparotomy and resection of the inflamed part of the bowel. We report a child with GIB who was successfully diagnosed endoscopically without surgical intervention.

Gastrointestinal basidiobolomycosis (GIB) is a rare fungal infection caused by basidiobolus ranarum. It has been recognized increasingly in the southern region of Saudi Arabia. Here, we report an unusual case of pediatric GIB with anorectal involvement in a 7-year-old Yemeni boy living in the south of Saudi Arabia. He had abdominal pain, abdominal distension, rectal bleeding, weight loss, perianal swelling and redness, and peripheral eosinophilia. The abdominal computed tomographic (CT) scan revealed significant wall thickness of the rectum. Histopathological findings of full thickness biopsy were consistent with basidiobolomycosis. The patient was treated with voriconazole and showed marked improvement. This case highlights the importance of considering GIB in children presenting with bleeding per rectum and eosinophilia associated with anorectal mass.

IntroductionBasidiobolomycosis is a rare fungal infection caused by Basidiobolus ranarum, and its incidence is poorly understood due to its rarity. Gastrointestinal Basidiobolomycosis (GIB) is an uncommon presentation with limited reports in Saudi Arabia. This retrospective study aims to describe risk factors, clinical findings, diagnosis, and management of GIB in the pediatric population.MethodsWe conducted a retrospective analysis of all pediatric patients diagnosed with GIB at a tertiary hospital between 2010 and 2022. Patients were identified based on their diagnosis of GIB. A self-administered questionnaire was distributed to assess risk factors associated with GIB.ResultsWe analyzed 25 cases of pediatric GIB, with approximately half of the cases (56%) occurring during the COVID-19 pandemic. The mean age of affected patients was 6.6 years, with a male predominance (60%, n=15). About 100% of parents acknowledged gecko existence in their residence, and their children were exposed to insect bites. Poor appetite was reported in all patients, and high counts of eosinophils and erythrocyte sedimentation rate were observed. Thrombocytosis was present in approximately 90% of patients, and 87% had anemia. Abdominal computerized tomography scans and ultrasound-guided biopsies were the most used diagnostic tools, with cases being diagnosed based on histopathological findings and confirmed via fungal culture.ConclusionOur study provides valuable information on the potential risk factors, clinical features, diagnosis, and management of pediatric GIB in the Jazan region. The observed increase in cases during the COVID-19 pandemic highlights the need for continued surveillance and research to understand better the epidemiology and associated risk factors with this rare fungal infection. These findings emphasize the need for heightened awareness, early detection, and effective prevention strategies to mitigate the impact of pediatric GIB.

-Basidiobolomycosis is a rare disease caused by the fungus Basidiobolus ranarum, an environmental saprophyte found worldwide. Patients with B ranarum infection may present with subcutaneous, gastrointestinal, or systemic lesions. Gastrointestinal basidiobolomycosis poses diagnostic difficulties, as its clinical presentation is nonspecific, with no identifiable risk factors. -To discuss and compare the clinical features and histopathologic findings and other ancillary techniques that could be helpful in identifying gastrointestinal basidiobolomycosis. -We report 3 cases of gastrointestinal basidiobolomycosis and describe the clinical and morphologic findings while emphasizing the importance of identifying this unusual entity on endoscopic biopsies, thus avoiding unnecessary major surgeries. Fungal cultures were also performed, which are of diagnostic significance. Our first patient was lost to follow-up; however, patients 2 and 3 were followed up for 4 and 2 years, respectively. -In all 3 cases, patients presented with a clinical profile suggestive of malignancy. None of the patients gave any specific history. There was widespread abdominal disease with peritoneal involvement and colonic masses. Colonoscopic biopsy specimens showed nonspecific inflammation in 1 case; however, they showed only granulomatous inflammation in a second case and granulomas associated with fungal hyphae in a third. Typical morphology included hyphae, irregularly branched, thin-walled, occasionally septated and surrounded by a thick eosinophilic cuff (Splendore-Hoeppli phenomenon). -Gastrointestinal basidiobolomycosis can be detected on small endoscopic biopsy. The unequivocal diagnosis requires microbiologic cultivation of the fungus obtained from tissues. The prognosis for this disease is usually favorable as seen in 3 of our cases; however, cases with fatal outcome are on record.

Basidiobolus ranarum is an environmental saprophyte found worldwide. It is a member of the order Entomophthorales of the class zygomycete. The infection is generally restricted to subcutaneous tissues. However, the disease can involve visceral organs, causing gastrointestinal basidiobolomycosis (GIB). GIB usually has nonspecific clinical manifestations and its diagnosis is challenging. Missed and delayed diagnosis of GIB increases the risk of morbidity and mortality, especially in pediatric patient. Previously, most of the cases in Saudi Arabia have been reported from southern region (Tohama, Aseer Region); the current study reports cases from different regions. We report a case of severe GIB and four other cases of pediatric GIB in western region of Saudi Arabia. The aim of our report is to describe the wide range of clinical presentations, diagnosis, management and outcomes. Our case series includes the youngest case report of GIB, a 16-month-old infant and highlights two important points. First, the need of high index of suspicious for diagnosis of GIB and its inclusion in the differential diagnosis of any abdominal mass, fever and eosinophilia. Second, it gives additional supportive evidence that medical management with voriconazole without surgical intervention (intervention if the condition is diagnosed early) is curative.

Gastrointestinal Basidiobolomycosis (GIB) is an unusual fungal disease caused by Basidiobolus ranarum, seen in the stomach, small intestine, colon, and liver. It has been rarely reported in the literature and most of the reported cases have been from US, Saudi Arabia, Kuwait, and Iran. Basidiobolus ranarum is an environmental saprophyte found worldwide. It is a member of the order Entomophthorales of the class zygomycete. The infection is generally restricted to subcutaneous tissues but the disease can also involve visceral organs.GIB usually has nonspecific clinical manifestations and its diagnosis is more challenging. Delayed diagnosis of GIB increases the risk of morbidity and mortality, especially in pediatric patient. The plan of our case report is to illustrate the wide range of clinical presentations, clinico-radiological and histopathological diagnosis, management and outcomes. Keywords: Basidiobolomycosis, Gastrointestinal, Pediatrics, Malignancy, Entomophthorales, Immunocompetent.

Background: Basidiobolomycosis is a fungal infection caused by Basidiobolus ranarum. It is primarily located in the tropical areas. It is commonly affecting the gastrointestinal tract and rarely presents with systemic affection.Patients and Methods: The records of fifteen patients, including pediatrics, from various geographic regions (western and southern regions of the Kingdom of Saudi Arabia) who were diagnosed with basidiobolomycosis in two tertiary hospitals over the last fourteen consecutive years from January 2010 to February 2023 were included. A detailed history and data for each patient were reviewed and evaluated.Results: A total of fifteen patients were found to have basidiobolomycosis, of which fourteen were found to have gastrointestinal basidiobolomycosis (GIB). They presented with abdominal pain, fever, nausea, vomiting, diarrhea, and loss of weight. One of them with an orbital infection presented with periorbital swelling, hotness, and redness. The diagnosis was confirmed after histopathological examination with typical features of granulomatous reaction, dense infiltrate of eosinophils, and fungal structures. Seven patients were treated with antifungals alone, such as voriconazole or itraconazole. Eight patients were treated by surgical resection followed by antifungal medication. Ten patients demonstrated good clinical response and completed their treatment course, while one patient showed a slight improvement and is still receiving treatment and follow‐up. There have been four reported cases of death. The length of the therapy was scheduled based on how the patients responded clinically to the intervention.Conclusions: We present the series with the most patients collected to date, and it is shown that GIB is often misdiagnosed as cancer or inflammatory bowel disease. Histological analysis confirms the diagnosis of basidiobolomycosis. The choice of therapy is still up for debate: medical intervention alone or surgical resection followed by antifungal medication.

BackgroundPediatric gastrointestinal basidiobolomycosis is an unusual fungal infection caused by Basidiobolus ranarum, an environmental saprophyte found worldwide. Typically, basidiobolomycosis presents as a subcutaneous infection or soft tissue tumor-like lesion, and rarely involves the gastrointestinal tract. Gastrointestinal basidiobolomycosis is most common in young infants. It has no definitive clinical presentation, and almost all cases are misdiagnosed during the initial presentation.Case presentationWe report the case of a 4-year-old Saudi boy who presented to the emergency department with abdominal pain, nausea, vomiting, and weight loss. Ultrasonography revealed a target sign. Based on the ultrasonography findings, surgery was performed, which revealed the presence of intussusception. Eventually, the patient was diagnosed with intussusception secondary to intra-abdominal basidiobolomycosis based on the histological findings. The patient was readmitted and intravenous voriconazole therapy was initiated. One week after the second admission, the patient developed abdominal pain, nausea, vomiting, inability to hold down food, and constipation. Computed tomography of the abdomen was suggestive of small bowel obstruction, which was managed conservatively. The patient responded well and was subsequently discharged with a prescription of oral voriconazole.ConclusionsThis case reveals that gastrointestinal basidiobolomycosis can cause intussusception. This report will inform clinicians of the importance of considering gastrointestinal basidiobolomycosis in the differential diagnosis of chronic abdominal pain in children, even in the absence of fever or a clinically obvious abdominal mass, especially in countries such as Saudi Arabia, where cases have been reported.

Basidiobolus ranarum is a known cause of chronic subcutaneous zygomycosis. During the past decade, many cases have been reported with extracutaneous basidiobolomycosis. We aimed to review the medical literature on gastrointestinal basidiobolomycosis (GIB) as an emerging fungal infection causing a serious, and occasionally fatal, paediatric disease. We reviewed all reported cases of visceral basidiobolomycosis with special focus on the gastrointestinal involvement in children. Twenty-six cases of GIB have been reported worldwide, in 14 adults and 12 children. All cases presented with fever, abdominal pain with mass and high blood eosinophilia and were misdiagnosed as other chronic granulomatous diseases or malignancies. A few cases of retroperitoneal, pulmonary, nasal and disseminated basidiobolomycosis have also been reported. Basidiobolomycosis mostly affects young males as a subcutaneous infection. The visceral form of infection is rare. GIB has been scarcely reported in the medical literature, but recently it has been increasingly recognized. GIB poses diagnostic difficulties. Abdominal pain with mass and eosinophilia were present in all cases, highlighting the necessity of considering GIB in the differential diagnosis of this presentation. B. ranarum has been reported to cause disseminated fatal disease in both immune-competent and -compromised individuals. Culture is the gold standard for diagnosis, but the characteristic histopathological picture of chronic granulomas rich in eosinophils and the Splendore-Hoeppli phenomenon are the usual diagnostic tools. Surgery plus long courses of itraconazole treatment, up to 1 year, appear to be the best management options. GIB is an emerging infection that might lead to diagnostic confusion, morbidity and mortality. Diagnosis requires a high index of suspicion in the differential diagnosis of patients with fever, abdominal pain with mass and eosinophilia.

BackgroundBasidiobolomycosis is a rare disease caused by the fungus Basidiobolus ranarum, member of the class Zygomycetes, order Entomophthorales, found worldwide. Usually basidiobolomycosis is a subcutaneous infection but rarely gastrointestinal manifestations have been described; 13 adults and 10 children and a few retroperitoneal or pulmonary cases. In gastrointestinal basidiobolomycosis the colon is most frequently involved, usually presenting with subacute mild abdominal pain. In contrast to children only very few described adult patients had hepatic masses. Definitive diagnosis requires culture, serological testing can be helpful. The fungal morphology and the Splendore-Hoeppli phenomenon are characteristic histological features. There are no prominent risk factors. Usually surgery and prolonged antifungal therapy are required.Case presentationA 61 year old man presented with progressive left abdominal pain and constipation since a few months. Colonoscopy showed an obstructing tumour in the descending colon, and a hemicolectomy was performed. Histology showed inflammation, possibly caused by a fungal or parasitic infection, without definite identification of an organism. A few weeks postoperatively a CT scan made because of abdominal discomfort, revealed a livermass (6 cm). Treatment with metronidazole, directed against an amoebic liver abscess, was unsuccessful. He developed a marked eosinophilia (27.7%). A liver biopsy was performed and the patient was referred to a university hospital.A repeated CT scan showed a livermass of 9 cm diameter. Review of colon and liver biopsy samples showed extensive necrosis and histiocytes, multinucleated giant cells and numerous eosinophils. Grocott stained sections contained unusually large hyphae surrounded by strongly eosinophilic material in haematoxylin and eosin stained sections (Splendore-Hoeppli phenomenon). A presumptive diagnosis of Basidiobolus spp. infection was made and treated with amphotericin B (Itraconazol contra-indicated because of renal insufficiency). A few days later the patient died of a septic shock. After autopsy Basidiobolus ranarum was cultured from liver, gallbladder and colon.ConclusionOur patient died of gastrointestinal basidiobolomycosis with an obstructing colon tumour and a large hepatic mass. This was a rare presentation of basidiobolomycosis and the second fatal case described worldwide.

Gastrointestinal basidiobolomycosis (GIB) is a rare fungal infection caused by the Basidiobolus ranarum, and it possesses a significant challenge to diagnose it as it presents with non-specific symptoms that often mimic cancer. Herein, we report a case of GIB in a 51-year-old male from the central region of Saudi Arabia, a non-endemic region of GIB, which was initially misdiagnosed as colon cancer. A 51-year-old man presented with abdominal pain for two-months, non-bloody diarrhea, loss of appetite, and weight loss. Abdominal examination revealed a large mass measuring ~10x15cm. Radiological findings prompted the diagnosis of a colon mass, and the patient was surgically treated under that impression. Hemicolectomy and end colostomy with mucous fistula from distal sigmoid stump were done. Histopathology was consistent with GIB. The diagnosis of GIB presents a serious challenge and requires a high index of clinical suspicion.

Introduction:Gastrointestinal basidiobolomycosis (GIB) is an emerging fungal infection with a few cases reported worldwide. It is caused by Basidiobolus ranarum, which does not usually invade blood vessels and rarely disseminates.Case presentation:We present a rare case of GIB with hepatic dissemination in a 12-year-old Yemeni boy living in southwestern Saudi Arabia. The initial provisional diagnosis was intestinal lymphoma, and a right hemicolectomy was carried out, but histopathological assessment ruled out lymphoma and suggested intestinal tuberculosis. Two weeks after starting antituberculous medications, the patient was referred to our hospital because of fever and right upper abdominal discomfort. There was leukocytosis with marked eosinophilia, and a liver biopsy showed evidence of B. ranarum infection. A second opinion by histopathological examination of resected tissues diagnosed colonic basidiobolomycosis. The patient was treated successfully with itraconazole.Conclusion:GIB is an emerging disease in southwestern Saudi Arabia and should be considered in a patient with an abdominal mass and eosinophilia coming from this region. Persistent elevation of leukocytes and eosinophils after surgical resection of the affected tissue could be used as a predictor of fungal dissemination. Further research is needed for a better understanding of GIB.

Gastrointestinal basidiobolomycosis (GIB) is an invasive fungal infection affecting immunocompetent children and young adults. Most of the reported cases are from the southwestern region of Saudi Arabia; it has a non-specific presentation which can mimic inflammatory bowel disease, gastrointestinal tuberculosis, and colon cancer. GIB usually affects the colon and small intestine while disseminated GIB is very rare. Liver involvement was reported in only 5 cases. We report here the case of a 22-year-old Saudi girl living in Jizan (southern region of Saudi Arabia) presented with disseminated gastrointestinal intramural and liver abscesses which were successfully treated with the aspiration of liver abscess and oral antifungal therapy (itraconazole). GIB diagnosis need a high index of suspicion and should be considered in patients from the southwestern region of Saudi Arabia with gastrointestinal intramural and liver abscesses. GIB is an emerging infection that might lead to diagnostic confusion, morbidity, and mortality.

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

Gastrointestinal basidiobolomycosis (GIB) is unusual, rare, and emerging fungal infection. It is caused by Basidiobolus ranarum. Unlike other fungal infections, B ranarum affects immunocompetent individuals with potentially grave sequelae if unrecognized. GIB is difficult to be diagnosed due to unspecific clinical presentation. Diagnosis of basidiobolomycosis requires culture of B ranarum from tissue. Optimal management of GIB includes antifungal therapy alongside if necessary early surgery.