Hypereosinophilic Syndrome Presenting as Eosinophilic Cholangiopathy and Cystitis / 대한췌담도학회지

ABSTRACT

Eosinophilic cholangiopathy is a rare disease characterized by dense transmural eosinophilic infiltration of the gallbladder and bile duct. It's clinical and laboratory manifestations are not different from those of other causes of cholangiopathy, and the diagnosis is usually made based on pathologic findings after cholecystectomy. Moreover, the occurrence of eosinophilic cystitis accompanied by cholangiopathy is extremely rare. We report a rare case of hypereosinophilic syndrome manifested as eosinophilic cholangiopathy accompanied with eosinophilic cystitis, for the first time in Korea, in a 49-year-old woman who presented with persistent right upper quadrant pain. After performing imaging study to validate the diagnosis of acute acalculous cholecystitis and cholangitis, an urgent cholecystectomy was performed. Pathologic examination of the excised gallbladder was consistent with eosinophilic cholecystitis. The patient underwent bladder biopsy because there was persistant irritative voiding symptoms combined with constant mild peripheral eosinophilia even after cholecystectomy, and the pathologic findings revealed eosinophilic cystitis. Symptoms and peripheral eosinophilia were improved after steroid therapy for an indicated period.