Primary sclerosing cholangitis with partial steroid responsiveness: a case report
A 69-year-old woman suspected to have IgG4-related sclerosing cholangitis causing bile duct stenosis was transferred from another hospital after diarrhea, eosinophilia, and eosinophilic infiltration were detected and prednisolone was prescribed. Additional biliary imaging suggested primary sclerosin...
| Main Authors: | , , , , |
|---|---|
| Format: | Article |
| Language: | English |
| Published: |
Fujita Medical Society
2023-05-01
|
| Series: | Fujita Medical Journal |
| Subjects: | |
| Online Access: | https://www.jstage.jst.go.jp/article/fmj/9/2/9_2022-012/_pdf/-char/en |
| _version_ | 1797831734092365824 |
|---|---|
| author | Satoshi Yamamoto Kazuo Inui Yoshiaki Katano Hironao Miyoshi Kenji Notohara |
| author_facet | Satoshi Yamamoto Kazuo Inui Yoshiaki Katano Hironao Miyoshi Kenji Notohara |
| author_sort | Satoshi Yamamoto |
| collection | DOAJ |
| description | A 69-year-old woman suspected to have IgG4-related sclerosing cholangitis causing bile duct stenosis was transferred from another hospital after diarrhea, eosinophilia, and eosinophilic infiltration were detected and prednisolone was prescribed. Additional biliary imaging suggested primary sclerosing cholangitis, but the IgG4 level and inferior bile duct stenosis were alleviated by steroid therapy, suggesting IgG4-related sclerosing cholangitis. Therefore, prednisolone was continued. Bile duct biopsy findings suggesting adenocarcinoma led to a diagnosis of pancreatoduodenectomy. The latter specimen only displayed evidence of primary sclerosing cholangitis, and prednisolone was discontinued. Intractable cholangitis necessitated left hepatectomy, after which serum alkaline phosphatase levels increased and eosinophilic colitis recurred. The reintroduction of prednisolone effectively managed the diarrhea but only temporarily reversed the alkaline phosphatase elevation. When histologic sections from resection specimens were compared, the hepatectomy specimen exhibited greater eosinophil infiltration than the earlier pancreatoduodenectomy specimen, suggesting eosinophilic cholangiopathy superimposed on primary sclerosing cholangitis. |
| first_indexed | 2024-04-09T13:56:31Z |
| format | Article |
| id | doaj.art-215e2d10117f4d6e92fc0c35e081e5f1 |
| institution | Directory Open Access Journal |
| issn | 2189-7247 2189-7255 |
| language | English |
| last_indexed | 2024-04-09T13:56:31Z |
| publishDate | 2023-05-01 |
| publisher | Fujita Medical Society |
| record_format | Article |
| series | Fujita Medical Journal |
| spelling | doaj.art-215e2d10117f4d6e92fc0c35e081e5f12023-05-08T04:03:35ZengFujita Medical SocietyFujita Medical Journal2189-72472189-72552023-05-0192154159https://doi.org/10.20407/fmj.2022-012Primary sclerosing cholangitis with partial steroid responsiveness: a case reportSatoshi Yamamoto0Kazuo Inui1Yoshiaki Katano2Hironao Miyoshi3Kenji Notohara4Department of Gastroenterology, Fujita Health University Bantane HospitalDepartment of Gastroenterology, Yamashita HospitalDepartment of Gastroenterology, Fujita Health University Bantane HospitalDepartment of Gastroenterology, Fujita Health University Bantane HospitalDepartment of Anatomic Pathology, Kurashiki Central HospitalA 69-year-old woman suspected to have IgG4-related sclerosing cholangitis causing bile duct stenosis was transferred from another hospital after diarrhea, eosinophilia, and eosinophilic infiltration were detected and prednisolone was prescribed. Additional biliary imaging suggested primary sclerosing cholangitis, but the IgG4 level and inferior bile duct stenosis were alleviated by steroid therapy, suggesting IgG4-related sclerosing cholangitis. Therefore, prednisolone was continued. Bile duct biopsy findings suggesting adenocarcinoma led to a diagnosis of pancreatoduodenectomy. The latter specimen only displayed evidence of primary sclerosing cholangitis, and prednisolone was discontinued. Intractable cholangitis necessitated left hepatectomy, after which serum alkaline phosphatase levels increased and eosinophilic colitis recurred. The reintroduction of prednisolone effectively managed the diarrhea but only temporarily reversed the alkaline phosphatase elevation. When histologic sections from resection specimens were compared, the hepatectomy specimen exhibited greater eosinophil infiltration than the earlier pancreatoduodenectomy specimen, suggesting eosinophilic cholangiopathy superimposed on primary sclerosing cholangitis.https://www.jstage.jst.go.jp/article/fmj/9/2/9_2022-012/_pdf/-char/eneosinophiliaigg4-related sclerosing cholangitisprimary sclerosing cholangitiseosinophilic cholangiopathy |
| spellingShingle | Satoshi Yamamoto Kazuo Inui Yoshiaki Katano Hironao Miyoshi Kenji Notohara Primary sclerosing cholangitis with partial steroid responsiveness: a case report Fujita Medical Journal eosinophilia igg4-related sclerosing cholangitis primary sclerosing cholangitis eosinophilic cholangiopathy |
| title | Primary sclerosing cholangitis with partial steroid responsiveness: a case report |
| title_full | Primary sclerosing cholangitis with partial steroid responsiveness: a case report |
| title_fullStr | Primary sclerosing cholangitis with partial steroid responsiveness: a case report |
| title_full_unstemmed | Primary sclerosing cholangitis with partial steroid responsiveness: a case report |
| title_short | Primary sclerosing cholangitis with partial steroid responsiveness: a case report |
| title_sort | primary sclerosing cholangitis with partial steroid responsiveness a case report |
| topic | eosinophilia igg4-related sclerosing cholangitis primary sclerosing cholangitis eosinophilic cholangiopathy |
| url | https://www.jstage.jst.go.jp/article/fmj/9/2/9_2022-012/_pdf/-char/en |
| work_keys_str_mv | AT satoshiyamamoto primarysclerosingcholangitiswithpartialsteroidresponsivenessacasereport AT kazuoinui primarysclerosingcholangitiswithpartialsteroidresponsivenessacasereport AT yoshiakikatano primarysclerosingcholangitiswithpartialsteroidresponsivenessacasereport AT hironaomiyoshi primarysclerosingcholangitiswithpartialsteroidresponsivenessacasereport AT kenjinotohara primarysclerosingcholangitiswithpartialsteroidresponsivenessacasereport |