甲状腺激素β抵抗儿童的一种新的THRβ变异:诊断和治疗挑战

IF 1.5 4区 医学 Q4 ENDOCRINOLOGY & METABOLISM
Gözde Gürpınar, Duygu Gamze Aracı
{"title":"甲状腺激素β抵抗儿童的一种新的THRβ变异:诊断和治疗挑战","authors":"Gözde Gürpınar, Duygu Gamze Aracı","doi":"10.4274/jcrpe.galenos.2025.2025-6-7","DOIUrl":null,"url":null,"abstract":"<p><p>Resistance to thyroid hormone beta (RTHβ) is a rare condition typically caused by mutations in the <i>THRβ</i> gene, characterized by elevated thyroid hormones with non-suppressed TSH levels. We present a pediatric case of RTHβ associated with a novel <i>THRβ</i> variant, emphasizing diagnostic challenges and the importance of individualized treatment. A 6.5-year-old girl was evaluated for learning difficulties and tachycardia. Laboratory findings showed elevated free T3 and T4 with non-suppressed TSH. Pituitary MRI showed a 5x6 mm lesion, raising suspicion for TSHoma. Genetic testing of the <i>THRβ</i> gene was performed. A novel heterozygous <i>THRβ</i> variant (c.1376T>C; p.Phe459Ser) was identified in both the patient and her father. The mutation affects a highly conserved residue within the ligand-binding domain. Clinical and biochemical findings were consistent with RTHβ. Atenolol therapy was initiated to manage tachycardia with favorable response.This case highlights the potential for misdiagnosis of RTHβ as TSHoma and underscores the value of genetic testing in differentiating the two. The identification of a novel variant at codon 459 expands the mutational spectrum of <i>THRβ</i> and supports its role as a hotspot region relevant to RTHβ pathogenesis.</p>","PeriodicalId":48805,"journal":{"name":"Journal of Clinical Research in Pediatric Endocrinology","volume":" ","pages":""},"PeriodicalIF":1.5000,"publicationDate":"2025-09-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"A Novel <i>THRβ</i> Variant in a Child With Resistance to Thyroid Hormone β: Diagnostic and Therapeutic Challenges.\",\"authors\":\"Gözde Gürpınar, Duygu Gamze Aracı\",\"doi\":\"10.4274/jcrpe.galenos.2025.2025-6-7\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Resistance to thyroid hormone beta (RTHβ) is a rare condition typically caused by mutations in the <i>THRβ</i> gene, characterized by elevated thyroid hormones with non-suppressed TSH levels. We present a pediatric case of RTHβ associated with a novel <i>THRβ</i> variant, emphasizing diagnostic challenges and the importance of individualized treatment. A 6.5-year-old girl was evaluated for learning difficulties and tachycardia. Laboratory findings showed elevated free T3 and T4 with non-suppressed TSH. Pituitary MRI showed a 5x6 mm lesion, raising suspicion for TSHoma. Genetic testing of the <i>THRβ</i> gene was performed. A novel heterozygous <i>THRβ</i> variant (c.1376T>C; p.Phe459Ser) was identified in both the patient and her father. The mutation affects a highly conserved residue within the ligand-binding domain. Clinical and biochemical findings were consistent with RTHβ. Atenolol therapy was initiated to manage tachycardia with favorable response.This case highlights the potential for misdiagnosis of RTHβ as TSHoma and underscores the value of genetic testing in differentiating the two. The identification of a novel variant at codon 459 expands the mutational spectrum of <i>THRβ</i> and supports its role as a hotspot region relevant to RTHβ pathogenesis.</p>\",\"PeriodicalId\":48805,\"journal\":{\"name\":\"Journal of Clinical Research in Pediatric Endocrinology\",\"volume\":\" \",\"pages\":\"\"},\"PeriodicalIF\":1.5000,\"publicationDate\":\"2025-09-17\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Journal of Clinical Research in Pediatric Endocrinology\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.4274/jcrpe.galenos.2025.2025-6-7\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q4\",\"JCRName\":\"ENDOCRINOLOGY & METABOLISM\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal of Clinical Research in Pediatric Endocrinology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.4274/jcrpe.galenos.2025.2025-6-7","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q4","JCRName":"ENDOCRINOLOGY & METABOLISM","Score":null,"Total":0}
引用次数: 0

摘要

甲状腺激素β (RTHβ)抵抗是一种罕见的疾病,通常由THRβ基因突变引起,其特征是甲状腺激素升高而TSH水平未受抑制。我们报告了一例与一种新型THRβ变异相关的RTHβ儿科病例,强调了诊断挑战和个体化治疗的重要性。一个6.5岁的女孩被评估为学习困难和心动过速。实验室结果显示游离T3和T4升高,TSH未受抑制。垂体MRI示5x6mm病灶,怀疑为TSHoma。进行THRβ基因的基因检测。在患者及其父亲身上均发现了一种新的THRβ杂合变异(C . 1376t >C; p.Phe459Ser)。突变影响配体结合域内高度保守的残基。临床和生化结果与RTHβ一致。开始阿替洛尔治疗心动过速,反应良好。该病例强调了RTHβ误诊为TSHoma的可能性,并强调了基因检测在区分两者方面的价值。在密码子459上发现一个新的变异扩展了THRβ的突变谱,并支持其作为与RTHβ发病机制相关的热点区域的作用。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
A Novel THRβ Variant in a Child With Resistance to Thyroid Hormone β: Diagnostic and Therapeutic Challenges.

Resistance to thyroid hormone beta (RTHβ) is a rare condition typically caused by mutations in the THRβ gene, characterized by elevated thyroid hormones with non-suppressed TSH levels. We present a pediatric case of RTHβ associated with a novel THRβ variant, emphasizing diagnostic challenges and the importance of individualized treatment. A 6.5-year-old girl was evaluated for learning difficulties and tachycardia. Laboratory findings showed elevated free T3 and T4 with non-suppressed TSH. Pituitary MRI showed a 5x6 mm lesion, raising suspicion for TSHoma. Genetic testing of the THRβ gene was performed. A novel heterozygous THRβ variant (c.1376T>C; p.Phe459Ser) was identified in both the patient and her father. The mutation affects a highly conserved residue within the ligand-binding domain. Clinical and biochemical findings were consistent with RTHβ. Atenolol therapy was initiated to manage tachycardia with favorable response.This case highlights the potential for misdiagnosis of RTHβ as TSHoma and underscores the value of genetic testing in differentiating the two. The identification of a novel variant at codon 459 expands the mutational spectrum of THRβ and supports its role as a hotspot region relevant to RTHβ pathogenesis.

求助全文
通过发布文献求助,成功后即可免费获取论文全文。 去求助
来源期刊
Journal of Clinical Research in Pediatric Endocrinology
Journal of Clinical Research in Pediatric Endocrinology ENDOCRINOLOGY & METABOLISM-PEDIATRICS
CiteScore
3.60
自引率
5.30%
发文量
73
审稿时长
20 weeks
期刊介绍: The Journal of Clinical Research in Pediatric Endocrinology (JCRPE) publishes original research articles, reviews, short communications, letters, case reports and other special features related to the field of pediatric endocrinology. JCRPE is published in English by the Turkish Pediatric Endocrinology and Diabetes Society quarterly (March, June, September, December). The target audience is physicians, researchers and other healthcare professionals in all areas of pediatric endocrinology.
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:604180095
Book学术官方微信