Chen YH, Chen QQ, Wang CL. Treatment and five-year follow-up of type A insulin resistance syndrome: A case report. World J Clin Cases 2022; 10(8): 2522-2528 [PMID: 35434067 DOI: 10.12998/wjcc.v10.i8.2522]
Corresponding Author of This Article
Chun-Lin Wang, Doctor, PhD, Chief Doctor, Professor, Department of Pediatrics, the First Affiliated Hospital, Zhejiang University School of Medicine, No. 79 Qingchun Road, Shangchcheng District, Hangzhou 310003, Zhejiang Province, China. hzwangcl@zju.edu.cn
Research Domain of This Article
Endocrinology & Metabolism
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. Mar 16, 2022; 10(8): 2522-2528 Published online Mar 16, 2022. doi: 10.12998/wjcc.v10.i8.2522
Treatment and five-year follow-up of type A insulin resistance syndrome: A case report
Yong-Hua Chen, Qing-Qing Chen, Chun-Lin Wang
Yong-Hua Chen, Qing-Qing Chen, Chun-Lin Wang, Department of Pediatrics, the First Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou 310003, Zhejiang Province, China
Author contributions: Wang CL conceived and designed the research; Chen YH and Chen QQ analyzed the data. Chen YH, Chen QQ, and Wang CL wrote the manuscript.
Informed consent statement: Informed written consent was obtained from the patient’s parents for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflicts of interest related to this manuscript.
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: Chun-Lin Wang, Doctor, PhD, Chief Doctor, Professor, Department of Pediatrics, the First Affiliated Hospital, Zhejiang University School of Medicine, No. 79 Qingchun Road, Shangchcheng District, Hangzhou 310003, Zhejiang Province, China. hzwangcl@zju.edu.cn
Received: July 27, 2021 Peer-review started: July 27, 2021 First decision: October 22, 2021 Revised: November 2, 2021 Accepted: February 10, 2022 Article in press: February 10, 2022 Published online: March 16, 2022 Processing time: 226 Days and 13 Hours
Abstract
BACKGROUND
Type A insulin resistance syndrome (TAIRS) is a rare disorder characterized by severe insulin resistance due to defects in insulin receptor signaling. No specific drugs are available for the treatment of TAIRS. We report a case of TAIRS successfully treated with pioglitazone and flutamide for 5 years.
CASE SUMMARY
We present the rare case of a female patient aged 11 years and 9 mo with type A insulin resistance and an INSR heterozygous mutation (c.3614C>T), who was treated with a combination of pioglitazone and flutamide. This treatment regimen reduced hemoglobin A1c, fasting insulin and androgen levels.
CONCLUSION
Pioglitazone attenuated insulin resistance in this patient with TAIRS, and flutamide ameliorated masculinization.
Core Tip: Type A insulin resistance syndrome (TAIRS) is a rare disorder characterized by severe insulin resistance due to defects in signaling through the insulin receptor. We present the rare case of a female patient aged 11 years and 9 mo who had type A insulin resistance with an INSR heterozygous mutation (c.3614C>T). This is the first case report describing the use of pioglitazone and flutamide used in combination in a child with TAIRS. This protocol for TAIRS is inexpensive, effective, and free of side effects.
