Journal of Pathology and Translational Medicine

Case Study

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; DOI: https://doi.org/10.4132/jptm.2015.12.01

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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

Citations to this article as recorded by

Pathologic interpretation of endoscopic ultrasound–guided fine needle aspiration cytology/biopsy for pancreatic lesions
Haeryoung Kim, Kee-Taek Jang
Journal of Pathology and Translational Medicine.2020; 54(5): 367. CrossRef
Multivisceral IgG4-related disease presenting as recurrent massive gastrointestinal bleeding: a case report and literature review
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

Case Reports

Adenosquamous Carcinoma of the Ampulla of Vater: A Report of Two Cases.: Da keun Lee, Cheol Keun Park, Geung hwan Ahn, Sung Ju Kim, Jin Seok Heo, Kyu Taek Lee, Sang hui Park, Kee Taek Jang; Korean J Pathol. 2006;40(2):160-164.

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Abstract PDF: Primary adenosquamous carcinoma of the ampulla of Vater is extremely rare. We report two cases of adenosquamous carcinoma of the ampulla of Vater in a 48-year-old female and an 80-year-old female patient. The gross appearance was a polypoid mass in both cases. Histologically, the majority of the two tumors was composed of squamous cell carcinoma with some intermingled glandular adenocarcinoma components in both cases. Squamous cell carcinomas showed an abrupt transition from the normal glandular epithelium or glandular adenocarcinoma. Although the two cases revealed duodenal wall invasion and lymph node metastasis was found in case 2, both patients are still alive at 19 and 46 months after surgery, respectively.

Adenomyoma of Ampulla of Vater or the Common Bile Duct: A Report of Three Cases.: Kee Taek Jang, Jin Seok Heo, Seoung Ho Choi, Dong Il Choi, Jae Hoon Lim, Young Lyun Oh, Geung Hwan Ahn; Korean J Pathol. 2005;39(1):59-62.

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Abstract PDF: Adenomyoma is a rare non-neoplastic lesion of the biliary tract. Here we report on three cases of adenomyoma; one located in the ampulla of Vater and two located in the common bile duct. Although preoperative endoscopic and radiological evaluations could not determine whether lesions were benign or malignant, intra-operative frozen section histologic examinations aided the differential diagnosis. Microscopic features of a lobular gland architecture with basally located nuclei and the absence of desmoplastic stromal reaction were found to be characteristic in frozen and paraffin sections.

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