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Jin Seok Heo 1 Article
Isolated Mass-Forming IgG4-Related Cholangitis as an Initial Clinical Presentation of Systemic IgG4-Related Disease
Seokhwi Kim, Hyunsik Bae, Misun Choi, Binnari Kim, Jin Seok Heo, Ho Seong Kim, Seung Hee Choi, Kee-Taek Jang
J Pathol Transl Med. 2016;50(4):300-305.   Published online January 11, 2016
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AbstractAbstract PDF
IgG4-related disease (IgG4-RD) may involve multiple organs. Although it usually presents as diffuse organ involvement, localized mass-forming lesions have been occasionally encountered in pancreas. However, the same pattern has been seldom reported in biliary tract. A 61-year-old male showed a hilar bile duct mass with multiple enlarged lymph nodes in imaging studies and he underwent trisectionectomy under impression of cholangiocarcinoma. Gross examination revealed a mass-like lesion around hilar bile duct. Histopathologically, dense lymphoplasmacytic infiltration and storiform fibrosis were identified without evidence of malignancy. Immunohistochemical stain demonstrated rich IgG4-positive plasma cell infiltration. Follow-up imaging studies disclosed multiple enlarged lymph nodes with involvement of pancreas and perisplenic soft tissue. The lesions have been significantly reduced after steroid treatment, which suggests multi-organ involvement of systemic IgG4-RD. Here, we report an unusual localized mass-forming IgG4-related cholangitis as an initial presentation of IgG4-RD, which was biliary manifestation of systemic IgG4-related autoimmune disease.


Citations to this article as recorded by  
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    Haeryoung Kim, Kee-Taek Jang
    Journal of Pathology and Translational Medicine.2020; 54(5): 367.     CrossRef
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    Xuexue Deng, Ronghua Fang, Jianshu Zhang, Rongqiong Li
    BMC Gastroenterology.2018;[Epub]     CrossRef
  • Recent advances in understanding and managing IgG4-related disease
    Anna R. Wolfson, Daniel L. Hamilos
    F1000Research.2017; 6: 185.     CrossRef

JPTM : Journal of Pathology and Translational Medicine