Comprehensive treatment and a rare presentation of Cronkhite–Canada syndrome: Two case reports and review of literature

doi:10.4240/wjgs.v15.i11.2646

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World Journal of Gastrointestinal Surgery

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1948-9366

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Baishideng Publishing Group Inc, 7041 Koll Center Parkway, Suite 160, Pleasanton, CA 94566, USA

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World J Gastrointest Surg. Nov 27, 2023; 15(11): 2646-2656
Published online Nov 27, 2023. doi: 10.4240/wjgs.v15.i11.2646

Comprehensive treatment and a rare presentation of Cronkhite–Canada syndrome: Two case reports and review of literature

Yan-Qing Lv, Mei-Lan Wang, Tong-Yu Tang, Yu-Qin Li

Yan-Qing Lv, Department of Hepatobiliary and Pancreatic Medicine, The First Hospital of Jilin University, Changchun 130021, Jilin Province, China

Mei-Lan Wang, Department of Gastroenterology, Jilin Provincial People's Hospital, Changchun 130021, Jilin Province, China

Tong-Yu Tang, Yu-Qin Li, Department of Gastroenterology, The First Hospital of Jilin University, Changchun 130021, Jilin Province, China

Author contributions: Lv YQ collected and sorted out the cases, reviewed the literature, and wrote the manuscript; Wang ML reviewed the literature; Tang TY contributed to the content and editing of the manuscript; Li YQ reviewed and revised the manuscript; All authors have read and approved the final manuscript.

Supported by Jilin Provincial Science and Technology Department Project, No. 20200201343JC; and Science and Technology Development Program of Jilin Province, No. 20210402013GH.

Informed consent statement: This study was approved by the Ethics Committee of the First Bethune Hospital of Jilin University to waive informed written consent about personal and medical data collection.

Conflict-of-interest statement: The authors declare that they have no conflict of interest 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).

Corresponding author: Yu-Qin Li, MD, Chief Physician, Professor, Department of Gastroenterology, The First Hospital of Jilin University, No. 71 Xinmin Street, Changchun 130021, Jilin Province, China. liyuq@jlu.edu.cn

Received: August 15, 2023
Peer-review started: August 15, 2023
First decision: September 1, 2023
Revised: September 15, 2023
Accepted: October 17, 2023
Article in press: October 17, 2023
Published online: November 27, 2023
Processing time: 103 Days and 20.8 Hours

Abstract

BACKGROUND

Cronkhite–Canada syndrome (CCS) is a rare sporadic polyposis syndrome that presents with gastrointestinal and ectodermal symptoms in addition to nutritional deficiencies. CCS combined with hypothyroidism is an even rarer condition, with no standard treatment guidelines.

CASE SUMMARY

The present study described 2 patients with CCS: A 67-year-old woman with concomitant hypothyroidism and 68-year-old man treated with endoscopic mucosal resection (EMR). Both patients had multiple gastrointestinal symptoms and ectodermal changes, along with multiple gastrointestinal polyps. Microscopic examination showed that the mucosa in both patients was hyperemic and edematous, with pathologic examination showing distorted, atrophic, and dilated glands. Patient 1 had concomitant hypothyroidism and was treated with levothyroxine. Due to her self-reduction of hormone dose, her disease relapsed. Patient 2 underwent EMR, but refused further hormonal or biological treatments. Subsequently, he was treated with an oral Chinese medical preparation.

CONCLUSION

Pharmacotherapy can induce and maintain remission in CCS patients, with adjuvant EMR, long-term follow-up, and endoscopic surveillance being necessary.

Keywords: Cronkhite–Canada syndrome; Clinical features; Gastrointestinal polyps; Hypothyroidism; Case report

Core Tip: Cronkhite–Canada syndrome (CCS) is a rare, non-genetic syndrome characterized by ectodermal abnormalities and diffuse gastrointestinal polyps with protein loss. To date, 7 patients with CCS combined with hypothyroidism have been identified. We report two cases, one of which is combined with hypothyroidism. Through indexing and analyzing PubMed, Web of Science, and Embase databases, we summarized the clinical characteristics of CCS combined with hypothyroidism. Additionally, we concluded that pharmacotherapy can induce and maintain remission in CCS patients, with adjuvant endoscopic mucosal resection, long-term follow-up, and endoscopic surveillance being necessary.