Acute Acalculous Cholecystitis: A Rare Presentation of Autoimmune Hepatitis and Primary Sclerosing Cholangitis Overlap Syndrome

Feng Yin, Peter V. Draganov, Jinping Lai, Steven J. Hughes, Roberto J. Firpi-Morell, Xiuli Liu


Lymphoplasmacytic cholecystitis (LPC) is a unique form of chronic cholecystitis, and it has been reported in patients with medical conditions such as primary sclerosing cholangitis, inflammatory bowel disease, and autoimmune pancreatitis. We report a case of LPC in a 23-year-old male with a history of autoimmune hepatitis who presented to the hospital with 3-day duration of abdominal pain, nausea and vomiting. A clinical diagnosis of acute cholecystitis and sclerosing cholangitis was made, and the patient was treated with laparoscopic cholecystectomy. Histopathological examination of the gallbladder revealed marked diffuse mononuclear inflammatory infiltrate that was rich in IgG4-positive plasma cells and focal neutrophilic inflammation with erosion. These findings were consistent with active LPC. The patients post-operative endoscopic retrograde cholangiopancreatography performed 1 month after cholecystectomy revealed typical features of primary sclerosing cholangitis. This is the first report of acute acalculous cholecystitis as an initial presentation of autoimmune hepatitis and primary sclerosing cholangitis overlap syndrome.

J Med Cases. 2018;9(10):332-337


Lymphoplasmacytic cholecystitis; Autoimmune hepatitis; Primary sclerosing cholangitis; Overlap syndrome; IgG4

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