Irving Levine, MD1, Kelly Suchman, MD1, Tai-Ping Lee, MD2 1Northwell Health, Manhasset, NY; 2Sandra B. Center for Liver Diseases, New Hyde Park, NY
Introduction: Eosinophilic cholangitis (EoC) is a rare and benign bile duct injury caused by eosinophilic infiltration of the biliary tree. Nearly all reported cases of EoC developed biliary strictures and obstructive jaundice, and diagnosis was made after excluding malignant etiologies. We herein report a unique case of non-stricturing eosinophilic cholangitis associated with pancreatitis in the absence of peripheral eosinophilia.
Case Description/Methods: A previously healthy 40-year-old man presented to our institution with four days of right upper quadrant abdominal pain and fever, without jaundice, nausea, or vomiting On hospital presentation, patient was febrile. Laboratory tests showed a WBC count of 12.4 x 109 cells/L with normal absolute eosinophilic count total bilirubin (TBili) of 2.6 mg/dL, and lipase of 609 U/L. Abdominal CT was notable for a diffusely thickened common bile duct wall. Despite broad spectrum antibiotics, patient remained febrile with rising bilirubin. Autoimmune antibodies and immunoglobulins, including IgG4, were normal, and viral hepatitis serologies were negative for acute infection. MRCP showed minimal intrahepatic biliary ductal dilatation with normal tapering of the CBD to the level of the ampulla. Additionally, there was diffuse biliary enhancement along the central left bile duct, common hepatic duct and common bile duct, and no choledocholithiasis(Figure 1). Patient underwent EUS showing diffuse wall thickening of the CBD, a lobular pancreas, and a normal pancreatic duct. Repeat MRCP on hospital day 7 showed similar thickening of CBD but interval development of a diffusely enlarged pancreas with preservation of normal acinar architecture, concerning for pancreatitis. Liver biopsy demonstrated prominent eosinophilic inflammatory infiltrate (90 to greater than 100 eos/HPI) in the portal tracts, with admixed neutrophils, lymphocytes, plasma cells and macrophages. By immunohistochemistry, there were only rare IgG4 lymphocytes present in portal tracts. Patient was started on prednisone 40mg daily, with improvement in abdominal pain and liver chemistries.
Discussion: Here we report a unique case of non-stricturing EoC causing concomitant pancreatitis. EoC is a rare and benign bile duct injury. Uniform diffuse enhancement of the entire CBD differs from other etiologies of cholangiopathy, such as choledocholithiasis, bacterial cholangitis, or primary sclerosing cholangitis, aiding in the diagnosis.
Figure: Figure 1: Contrast enhanced coronal T1 weighted MR image demonstrating diffuse enhancement of the extrahepatic bile duct (red arrow).
Irving Levine indicated no relevant financial relationships.
Kelly Suchman indicated no relevant financial relationships.
Tai-Ping Lee indicated no relevant financial relationships.
Irving Levine, MD1, Kelly Suchman, MD1, Tai-Ping Lee, MD2. P2151 - Non-Stricturing Eosinophilic Cholangitis Associated With Pancreatitis: A Unique Cause of Cholangiopathy, ACG 2021 Annual Scientific Meeting Abstracts. Las Vegas, Nevada: American College of Gastroenterology.