Rong JM, Shi ML, Niu JK, Luo J, Miao YL. Thalidomide combined with endoscopy in the treatment of Cronkhite-Canada syndrome: A case report. World J Clin Cases 2022; 10(28): 10366-10374 [PMID: 36246833 DOI: 10.12998/wjcc.v10.i28.10366]
Corresponding Author of This Article
Ying-Lei Miao, PhD, Chief Physician, Department of Gastroenterology, The First Affiliated Hospital of Kunming Medical University, No. 295 Xichang Road, Kunming 650032, Yunnan Province, China. miaoyinglei@kmmu.edu.cn
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/
World J Clin Cases. Oct 6, 2022; 10(28): 10366-10374 Published online Oct 6, 2022. doi: 10.12998/wjcc.v10.i28.10366
Thalidomide combined with endoscopy in the treatment of Cronkhite-Canada syndrome: A case report
Jia-Mei Rong, Meng-Lin Shi, Jun-Kun Niu, Juan Luo, Ying-Lei Miao
Jia-Mei Rong, Meng-Lin Shi, Jun-Kun Niu, Juan Luo, Ying-Lei Miao, Department of Gastroenterology, The First Affiliated Hospital of Kunming Medical University, Kunming 650032, Yunnan Province, China
Author contributions: Rong JM and Shi ML contributed to manuscript writing and editing, and data collection; Niu JK and Luo J helped revise the manuscript; Miao YL reviewed and revised the entire manuscript; all authors read and approved the final manuscript.
Supported byNational Natural Science Foundation of China, No. 82170550.
Informed consent statement: A written informed consent was obtained from the patient for publication of this case report.
Conflict-of-interest statement: The authors have nothing to disclose.
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: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Ying-Lei Miao, PhD, Chief Physician, Department of Gastroenterology, The First Affiliated Hospital of Kunming Medical University, No. 295 Xichang Road, Kunming 650032, Yunnan Province, China. miaoyinglei@kmmu.edu.cn
Received: July 9, 2022 Peer-review started: July 9, 2022 First decision: August 1, 2022 Revised: August 5, 2022 Accepted: August 24, 2022 Article in press: August 24, 2022 Published online: October 6, 2022 Processing time: 79 Days and 21 Hours
Abstract
BACKGROUND
Cronkhite-Canada syndrome (CCS) is a rare non-hereditary disease with a poor prognosis and a mortality rate of up to 55%. Currently, there is no standard treatment for CCS. The department of gastroenterology of our hospital admitted a patient with CCS whose symptoms improved significantly after treatment with thalidomide combined with endoscopy, and there was no obvious adverse reaction during the 2-year follow-up.
CASE SUMMARY
A 47-year-old Chinese man presented with diarrhea for more than 4 mo, accompanied by loss of taste, fatigue, and weight loss. Physical examination demonstrated that the patient’s skin and hands were hyperpigmented, the front edges of the nails of both hands were notably thickened and yellow, and the nails were partially atrophied. Gastrointestinal endoscopy identified a diffuse polypoid bulge, and the patient bore an albumin level of 27.3 g/L. The level of the calcium correction amount was (2.164 mM) which allowed for a comprehensive diagnosis of Cronkhite-Canada syndrome, combined with hypoalbuminemia and hypocalcemia. Thalidomide of 150 mg per day was administered to regulate immunity, and the symptoms were relieved after 1 wk. During the follow-up period, polyps were still found that had not been resolved by thalidomide treatment, and endoscopic therapy was performed. This resulted in further improvement of his condition and no particular discomfort during the 2 years of follow-up.
CONCLUSION
The patient’s symptoms were significantly relieved by thalidomide 2 years after treatment, proposing it as a potential treatment for CCS.
Core Tip: Cronkhite-Canada syndrome (CCS) is a rare non-hereditary disease with a poor prognosis and a mortality rate of up to 55%. Currently, there is no standard treatment for CCS. The symptoms of the patient in this case were significantly improved after treatment with thalidomide combined with endoscopy, and they were followed up for 2 years. No obvious adverse reactions were observed. Thalidomide may be a new potential therapeutic drug for CCS, and we will continue to follow up to determine its long-term efficacy.
