[Intraductal papillary neoplasm of bile duct developed in a patient with IgG4-related sclerosing cholangitis, autoimmune pancreatitis, and myasthenia gravis]

Nihon Shokakibyo Gakkai Zasshi. 2019;116(5):443-451. doi: 10.11405/nisshoshi.116.443.
[Article in Japanese]

Abstract

In the course of treatment for myasthenia gravis, enlargement of a cystic mass in the liver with peripheral bile duct dilation, diffuse pancreatic enlargement, and serum IgG4 level elevation was identified in a 65-year-old man. Following the diagnosis of autoimmune pancreatitis, a left hepatectomy was performed because of suspected malignancy of the cystic lesion. Analysis of the resected specimen revealed the cystic lesion to be a dilated bile duct. Intraductal papillary tumor comprising fibrovascular stalks covered by neoplastic epithelium was identified in the lesion. Infiltration of IgG4-positive plasma cells was discovered around the cystic lesion. Finally, a diagnosis of intraductal papillary neoplasm of bile duct with IgG4-related sclerosing cholangitis was made. Autoimmune diseases, including IgG4-related diseases, require careful observation because of their potential for malignancy.

MeSH terms

  • Aged
  • Autoimmune Diseases / complications
  • Autoimmune Diseases / diagnosis*
  • Bile Ducts
  • Cholangitis, Sclerosing / complications
  • Cholangitis, Sclerosing / diagnosis*
  • Humans
  • Immunoglobulin G / metabolism*
  • Male
  • Myasthenia Gravis / complications
  • Myasthenia Gravis / diagnosis*
  • Pancreatitis / complications
  • Pancreatitis / diagnosis*

Substances

  • Immunoglobulin G