{"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}
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.
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.