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Gastrointestinal Basidiobolomycosis: an Emerging Fungal Infection of The CASE FILES Gastrointestinal Basidiobolomycosis: An Emerging Fungal Infection of the Gastrointestinal Tract, the Royal Hospital (Sultanate of Oman) Experience Mohammed Al-Masqari1, Amal Al-Maani2, Fatma Ramadhan3 ABSTRACT Aims and objectives: In this study, we aimed to examine the epidemiology, clinical characteristics, histopathology findings, management, and ancillary techniques which are important for diagnosis, management, and outcome of an uncommon manifestation caused by the Basidiobolus ranarum. Materials and methods: This is a case series of five interesting cases of gastrointestinal basidiobolomycosis (GIB). This study was conducted in the histopathology department at the Royal hospital which is a tertiary care institution. Cases diagnosed with all types of fungal gastrointestinal disease between 2008 and 2015 were reviewed. Cases with morphological features of basidiobolomycosis were retrieved and the diagnosis was confirmed by a senior pathologist. Results: Five cases were identified. Out of five patients, four were misdiagnosed with other types of fungal infections which resulted in high morbidity and mortality. This case series revealed that the majority of patients identified are pediatrics (60%). Further, (60%) were from the same region (Ad Dakhiliyah). All patients presented with unspecific gastrointestinal symptoms that clinically mimic serious diseases. Additionally, all patients shared similar radiological findings and laboratory investigations. Conclusion: Diagnosis of GIB requires a high index of suspicion, increased awareness of this rare disease aid in early diagnosis and promote an early start of treatment. Since there is a resemblance in the clinical features of inflammatory and neoplastic bowel disease, GIB should be considered in the differential diagnosis. Keywords: Fungal infectious, Gastroenterology, Gastrointestinal basidiobolomycosis, Microbiology, Pathology. Pediatric Infectious Disease (2021): 10.5005/jp-journals-10081-1290 INTRODUCTION 1,3Department of Histopathology, The Royal Hospital, Muscat, Basidiobolomycosis is a rare fungal disease caused by Basidiobolus Sultanate of Oman ranarum, a fungus belonging to the family of Entomophthoraceae of 2Department of Pediatrics, The Royal Hospital, Muscat, Sultanate of the class Zygomycetes. It was first described by Eidam was the first Oman to describe Basidiobolomycosis as an isolate from frogs in 1886 and Corresponding Author: Mohammed Al-Masqari, Department of was later cultured from intestinal contents and the excreta of frogs.1 Histopathology, The Royal Hospital, Muscat, Sultanate of Oman, Infection by this fungus rarely affects the gastrointestinal tract but Phone: +968-24599617, e-mail: [email protected] mainly causes cutaneous disease involving the limbs, trunk, and How to cite this article: Al-Masqari M, Al-Maani A, Ramadhan F. buttocks.1 It is presumed that infection is acquired after minor Gastrointestinal Basidiobolomycosis: An Emerging Fungal Infection trauma to the skin or insect bites. The causative agent is common of the Gastrointestinal Tract, the Royal Hospital (Sultanate of Oman) in soil, decaying vegetable matter, and the dung of amphibians, Experience. Pediatr Inf Dis 2021;3(1):46–49. reptiles, fish, and insectivorous. Source of support: Nil Lack of awareness of gastrointestinal basidiobolomycosis (GIB) Conflict of interest: None has resulted in its delayed diagnosis and extensive morbidity and mortality in patients with GIB. Awareness of this disease will result in early diagnosis and hence will prevent unfavorable outcomes and were reviewed by a histopathology resident and a senior such as a reduction in unnecessary surgeries and likely reduction consultant pathologist. in mortality.2 This research will be the first and the largest case The cases of GIB are identified based on the characteristic serious to be done in Oman and will add to the insufficient literature histopathologic appearance of broad, thin-walled, pleomorphic published globally. hyphae surrounded by an eosinophilic material (known as the In this study, we aimed to investigate the epidemiology, clinical Splendore–Hoeppli phenomenon) (Fig. 1). Because this is a rare characteristics, histopathology findings, management, and ancillary disease; a limited number of studies were conducted including 3–5 techniques which are important for diagnosis, management, and small sample sizes. Hence, the sample size (n = 5) for this research outcome of an uncommon manifestation caused by the B. ranarum. is deemed acceptable. Data collected included demographic information, clinical presentation, abnormal laboratory findings, MATERiaLS AND METHODS sites of involvement, radiologic studies, treatment, and outcome. All the cases of GI fungal infection from 2008 to 2015 were identified Data were revised by a senior pathologist and an infectious disease through the medical record of the Royal Hospital (Al-Shifa System) consultant before the analysis. This study has been approved by the © Jaypee Brothers Medical Publishers. 2021 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (https://creativecommons.org/licenses/by-nc/4.0/), which permits unrestricted use, distribution, and non-commercial reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Gastrointestinal Basidiobolomycosis diagnosis is GIB. this case was misdiagnosed when mucormycosis as the actual metabolic acidosis, and hyperkalemia. This study revealed that patient died due severe to sepsis, disseminated fungal infection, with antibiotics and antifungal (Caspofungin, The Ambisome). intestinal, large intestinal wall thickening. The patient was treated was CT mucormycosis. abdomen showed diffuse gastric, small hyphae within pink eosinophilic materials, and the given diagnosis infiltration by lymphocyte and eosinophils, thick branching fungal mass was biopsied and the histopathological examinations showed anemia, mild leukocytosis, thrombocytosis, The chest and high CRP. mass. Laboratory investigations showed microcystic hypochromic impression was pneumonia the as chest showed X-ray alarge chest pain, productive cough, The vomiting, first and fever. clinical A 19-year-old girl presented the to Royal Hospital with abdominal 2 Case misdiagnosedwhen mucormycosis as the actual diagnosis GIB. is and multi-organ failure. This study revealed this case that was time. The patient died due severe to fungal sepsis, septic shock, IV voriconazole was not started it as was not available that at was mucormycosis. The patient was treated with antibiotics but Splendore–Hoepplidiagnosis the by phenomenon. The final eosinophils andmany some large fungal hyphae surrounded which showed adense inflammatory infiltrate composed of abscess. The tissue was sent for histopathologicalexamination remove the inflammatory mass which intraoperatively showed to done was Laparotomy process. neoplastic for suggestive a large loculated mass in the right iliac fossa. The findings were was referred the to Royal Hospital where CT abdomen showed post-surgery, the patient developed severe abdominal pain and thrombocytosis, few A weeks protein and (CRP). high C-reactive showed microcystic hypochromic anemia, mild leukocytosis, significantpathological changes. Laboratory investigations which the patient had an appendectomy which showed no and diarrhea. This was diagnosed asacute appendicitis after abdominal pain, abdominal mass associated with vomiting A 9-year-old boy presented the to regional hospital with 1 Case C of Health, Sultanate of Oman 1). (Table Clinical Research Ethics Committee of the Royal Hospital, Ministry (knownmaterial astheSplendore-Hoeppli phenomenon) 1: Fig. A SE Broad, thin-walled hyphae surrounded by a collar ofeosinophilic acollar by surrounded hyphae thin-walled Broad, D ESCR I PT I ONS Pediatric Infectious Disease, Volume 3 Issue 1 (January–March 2021) Volume 3Issue1(January–March Disease, Infectious Pediatric Table 1: Summary of demographic information, clinical/radiological data and outcome Cases number Ages/years Gender Region Clinical presentation Clinical diagnosis Radiological findings Treatment Outcome 1 9 Male Ad Dakhiliyah Abdominal pain, Acute appendicitis Loculated abscess in the Antibiotics Died due to severe vomiting, diarrhea right iliac fossa fungal sepsis 2 19 Female Al Dhahirah Abdominal pain, Pneumonia Diffuse gastric, small Antibiotics Severe sepsis, productive cough, intestinal, large intestinal and antifungal disseminated fungal vomiting, and fever wall thickening (Caspofungin, infection, metabolic Ambisome) acidosis, and hyper- kalemia 3 45 Male Ad Dakhiliyah Abdominal pain, fever, Malignant colonic Diffuse irregular Right Died due to severe diarrhea, and marked tumor thickening of the cecum, hemicolectomy, fungal sepsis and weight loss ascending colon, and antibiotics, and metabolic acidosis transverse antifungal (Ambi- some) 4 5 Female Muscat Abdominal pain, Acute appendicitis Diffuse irregular Right Improved and abdominal mass, thickening of the right hemicolectomey, IV discharged from the vomiting, and fever side of the colon voriconazole hospital 5 10 Male Ad Dakhiliyah Abdominal pain, and loss Colonic intussuscep- Ileocecal mass
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