IgG4-related Sclerosing Cholangitis Complicated with Cholangiocarcinoma and Detected by Forkhead Box P3 Immunohistochemical Staining

Intern Med. 2021 Mar 15;60(6):859-866. doi: 10.2169/internalmedicine.5920-20. Epub 2020 Oct 21.

Abstract

An 80-year-old man was admitted due to biliary stricture with autoimmune pancreatitis. Although radiographical examinations suggested Immunoglobulin G4-related sclerosing cholangitis (IgG4-SC), punched biopsies from the bile duct revealed adenocarcinoma. In the resected specimen, abundant N-terminus of Forkhead box P3 (Foxp3)-positive cells were localized in cholangiocarcinoma (CCA) tissue, while IgG4-positive cells were spread around the entire bile duct. Therefore, the case was diagnosed with IgG4-SC accompanied by CCA, not sporadic CCA. We herein report an informative case wherein IgG4-positive cells were abundant in CCA tissue and Foxp3 immunohistochemical staining allowed us to determine that this case had two entities.

Keywords: autoimmune pancreatitis; cholangiocarcinoma; forkhead box P3; immunoglobulin G4-related disease; immunoglobulin G4-related sclerosing cholangitis.

Publication types

  • Case Reports

MeSH terms

  • Aged, 80 and over
  • Autoimmune Diseases* / diagnosis
  • Bile Duct Neoplasms* / complications
  • Bile Duct Neoplasms* / diagnosis
  • Bile Ducts, Intrahepatic
  • Cholangiocarcinoma* / complications
  • Cholangiocarcinoma* / diagnosis
  • Cholangitis, Sclerosing* / complications
  • Cholangitis, Sclerosing* / diagnosis
  • Diagnosis, Differential
  • Humans
  • Immunoglobulin G
  • Male
  • Staining and Labeling

Substances

  • Immunoglobulin G