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

Abstract

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. 1 MDP and VDC contributed equally to this work.

Gastrointestinal basidiobolomycosis: Beware of the great masquerade a case report

“…In a recent review of 102 cases of GIB, abdominal pain (86.3%) was the most common presenting symptom followed by weight loss (33.3%), abdominal distension (16.7%), vomiting (15.7%) and diarrhea (13.7%) with fever reported in 40.2% of patients and an abdominal mass was palpable in 30.4% of cases [10]. GIB most commonly involves the right colon [11] and frequently presents as abdominal mass mimicking colonic malignancy and inflammatory bowel disease [6].…”

Colonic basidiobolomycosis presenting with intestinal obstruction and a normal eosinophil count

“…Remarkably, our patient’s eosinophil count remained normal before and following surgery. Peripheral eosinophilia is observed in 76–94.0% of the cases [1,13,14]. Interestingly, the persistence of peripheral eosinophilia following surgery could indicate an on-going focus of infection and demand further investigation [1].…”

“…Optimal treatment of GIB requires combined early aggressive surgical intervention and prolonged use of antifungals. A combination of surgery plus antifungal treatment was used in the majority (77.5%) of patients [14]. There is no current consensus on the antifungals to be used to treat this disease.…”

Invasive Basidiobolomycosis Presenting as Retroperitoneal Fibrosis: A Case Report

“…The first case of GIB was reported in a Nigerian child in 1964 [11]. Most of the cases reported have been from Saudi Arabia, and have occurred in a young age group [12]. Making the initial diagnosis is usually difficult and requires a high index of suspicion.…”

“…The diagnosis is difficult because of its non-specific clinical presentation, which is usually an insidious onset abdominal pain, fever, weight loss, diarrhea, or the presence of an abdominal mass. The most commonly affected abdominal organs are colon (84%), small bowel (32%), liver (21%), and rarely, other organs such as the stomach may be involved [7,12,15]. Moreover, concurrent subcutaneous and visceral basidiobolomycosis has been described in renal-transplanted adult patients [16].…”