Biliary Bipolaris: A Rare Case of Disseminated Fungal Infection in the Biliary Tract

Abstract

This is a case of a 39-year-old African American woman with no past medical history who presented with new onset seizures. Initial CT and MRI of the brain demonstrated bilateral temporal masses. Upon further questioning, the patient revealed she had a one week history of mild RUQ abdominal pain and laboratory tests showed an alkaline phosphatase of 1322, AST 96, ALT 236, and total Bilirubin of 1.3. Subsequent CT of the chest, abdomen, and pelvis demonstrated marked thickening of the CBD and CHD walls of up to 8 mm. ERCP was performed, with initial cholangiogram showing multiple irregular shaped filling defects within the entire biliary tree and CBD and CHD dilation of up to 10 mm. After biliary sphincterotomy was performed, large amounts of brown and green fragments, characteristic of mold, were seen exiting the bile duct and that were collected for culture. Choledochoscopy was then performed showing multiple fragments of mold filling the lumen of the CBD and CHD. The bile duct aspirate and CBD biopsies was positive for Bipolaris species. The patient then underwent resection of the right temporal lesion, with cranial biopsies revealing Bipolaris species infection of the brain as well. The patient was treated with IV ambisome and Voriconazole for a total of 6 months. Repeat ERCP two months later to remove a previously placed plastic stent showed near resolution of the biliary tract irregularities, with improvement in biliary dilation and normalization of liver function tests. A literature search suggest that this is the first case report of tissue proven Biloparis infection in the biliary tract in any patient type, immunocompromised and competent. Bipolaris species, are a group fungi commonly found in soil and plant material, and are present worldwide. Though they are recognized pathogens of plants, they rarely cause infections in animals and humans. When they have been present in human infection, they have typically been associated with immunosuppression sinusitis and allergic broncopulmonary disease. The literature suggest that amphotericin B alone or in combination with imidazoles is the recommend treatment. Due to the devastating consequences of disseminated Bipolaris, early diagnosis and treatment are essential.Figure 1Figure 2Figure 3