Zhou QY, Zhou WX, Sun XY, Wu B, Zheng WY, Li Y, Qian JM. Autoimmune enteropathy and primary biliary cholangitis after proctocolectomy for ulcerative colitis: A case report and review of the literature. World J Gastroenterol 2021; 27(29): 4929-4938 [PMID: 34447236 DOI: 10.3748/wjg.v27.i29.4929]
Corresponding Author of This Article
Yue Li, MD, Associate Professor, Department of Gastroenterology, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, No. 1 Shuaifuyuan, Dongcheng District, Beijing 100730, China. yuelee76@gmail.com
Research Domain of This Article
Gastroenterology & Hepatology
Article-Type of This Article
Case Report
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Qing-Yang Zhou, Wei-Yang Zheng, Yue Li, Jia-Ming Qian, Department of Gastroenterology, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing 100730, China
Wei-Xun Zhou, Department of Pathology, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing 100730, China
Xi-Yu Sun, Bin Wu, Department of General Surgery, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing 100730, China
Author contributions: Zhou QY drafted the manuscript; Zhou WX collected the pathological data and interpret the histological examination; Zhou QY, Zheng WY, and Li Y contributed to clinical data collection and follow up; Wu B and Sun XY performed the operation; Li Y and Qian JM critically revised the manuscript; all authors have read and approved the final manuscript.
Supported byChinese Academy of Medical Sciences Innovation Fund for Medical Sciences (CIFMS), No. 2017-I2M-3-017.
Informed consent statement: Written informed consent was obtained from the patient for publication of this case report.
Conflict-of-interest statement: Dr. Li Y reports grants from Chinese Academy of Medical Sciences Innovation Fund for Medical Sciences, during the conduct of the study.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Yue Li, MD, Associate Professor, Department of Gastroenterology, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, No. 1 Shuaifuyuan, Dongcheng District, Beijing 100730, China. yuelee76@gmail.com
Received: February 25, 2021 Peer-review started: February 25, 2021 First decision: April 18, 2021 Revised: April 29, 2021 Accepted: July 13, 2021 Article in press: July 13, 2021 Published online: August 7, 2021
Abstract
BACKGROUND
Autoimmune enteropathy (AIE) and primary biliary cholangitis (PBC) are both immune-mediated diseases. AIE or PBC complicated with ulcerative colitis (UC) are rare. There are no cases of AIE and PBC diagnosed after proctocolectomy for UC reported before, and the pathogenesis of these comorbidities has not been revealed.
CASE SUMMARY
A middle-aged woman diagnosed with UC underwent subtotal colectomy and ileostomy due to the steroid-resistant refractory disease, and a restorative proctectomy with ileal pouch-anal anastomosis and proximal neoileostomy was postponed due to active residual rectal inflammation in January 2016. A few months after the neoileostomy, she began to suffer from recurrent episodes of watery diarrhea. She was diagnosed with postcolectomy enteritis and stoma closure acquired a good therapeutic effect. However, her symptoms of diarrhea relapsed in 2019, with different histological features of endoscopic biopsies compared with 2016, which showed apoptotic bodies, a lack of goblet and Paneth cells, and villous blunting. A diagnosis of AIE was established, and the patient’s stool volume decreased dramatically with the treatment of methylprednisolone 60 mg/d for 1 wk and tacrolimus 3 mg/d for 4 d. Meanwhile, her constantly evaluated cholestatic enzymes and high titers of antimitochondrial antibodies indicated the diagnosis of PBC, and treatment with ursodeoxycholic acid (16 mg/kg per day) achieved satisfactory results.
CONCLUSION
Some immune-mediated diseases may be promoted by operation due to microbial alterations in UC patients. Continuous follow-up is essential for UC patients with postoperative complications.
Core Tip: This is the first case report of autoimmune enteropathy and primary biliary cholangitis complicated with ulcerative colitis, and the female patient in this case suffered from a tortuous process of diagnosis and treatment. It is speculated that a microbial shift and subsequent immune response are involved in the pathogenesis of these comorbidities, and surgery may facilitate the progression of coexisting diseases. Understanding the progression of these diseases may help with their early recognition and treatment.
