Literature DB >> 33333891

Hypercholesterolemia in Two Siblings with Resistance to Thyroid Hormones Due to Disease-Causing Variant in Thyroid Hormone Receptor (THRB) Gene.

Maja Pajek1, Magdalena Avbelj Stefanija2,3, Katarina Trebusak Podkrajsek2,3, Jasna Suput Omladic2,3, Mojca Zerjav Tansek2,3, Tadej Battelino2,3, Urh Groselj2,3.   

Abstract

Resistance to thyroid hormone beta (RTHβ) is a syndrome characterized by a reduced response of target tissues to thyroid hormones. In 85% of cases, a pathogenic mutation in the thyroid hormone receptor beta (THRB) gene is found. The clinical picture of RTHβ is very diverse; the most common findings are goiter and tachycardia, but the patients might be clinically euthyroid. The laboratory findings are almost pathognomonic with elevated free thyroxin (fT4) levels and high or normal thyrotropin (TSH) levels; free triiodothyronin (fT3) levels may also be elevated. We present three siblings with THRB mutation (heterozygous disease-variant c.727C>T, p.Arg243Trp); two of them also had hypercholesterolemia, while all three had several other clinical characteristics of RTHβ. This is the first description of the known Slovenian cases with RTHβ due to the pathogenic mutation in the THRB gene. Hypercholesterolemia might be etiologically related with RTHβ, since the severity of hormonal resistance varies among different tissues and hypercholesterolemia in patients with THRB variants might indicate the relatively hypothyroid state of the liver. We suggest that cholesterol levels are measured in all RTHβ patients.

Entities:  

Keywords:  RTHβ; THRB; hypercholesterolemia; hypothyroidism; resistance to thyroid hormones; thyroid; thyroid hormone receptor

Mesh:

Substances:

Year:  2020        PMID: 33333891      PMCID: PMC7765296          DOI: 10.3390/medicina56120699

Source DB:  PubMed          Journal:  Medicina (Kaunas)        ISSN: 1010-660X            Impact factor:   2.430


  22 in total

Review 1.  Resistance to thyroid hormone.

Authors:  R E Weiss; S Refetoff
Journal:  Rev Endocr Metab Disord       Date:  2000-01       Impact factor: 6.514

Review 2.  Role of thyroid receptor β in lipid metabolism.

Authors:  Camilla Pramfalk; Matteo Pedrelli; Paolo Parini
Journal:  Biochim Biophys Acta       Date:  2010-12-29

3.  A case of thyroid hormone resistance: a rare mutation.

Authors:  Ana Pires Gonçalves; José Maria Aragüés; Ema Nobre; Ana Paula Barbosa; Mario Mascarenhas
Journal:  Arq Bras Endocrinol Metabol       Date:  2014-12

Review 4.  Resistance to thyroid hormone.

Authors:  Kazumichi Onigata; Gabor Szinnai
Journal:  Endocr Dev       Date:  2014-08-29

5.  A de novo L330S point mutation in thyroid hormone receptor beta gene in a Thai female with resistance to thyroid hormone.

Authors:  S Ditudompo; B Ongphiphadhanakul; S Chanprasertyotin; R Rajatanavin
Journal:  Endocr J       Date:  1999-12       Impact factor: 2.349

6.  Search for abnormalities of nuclear corepressors, coactivators, and a coregulator in families with resistance to thyroid hormone without mutations in thyroid hormone receptor beta or alpha genes.

Authors:  S Reutrakul; P M Sadow; S Pannain; J Pohlenz; G A Carvalho; P E Macchia; R E Weiss; S Refetoff
Journal:  J Clin Endocrinol Metab       Date:  2000-10       Impact factor: 5.958

7.  Identification of four novel mutations in the thyroid hormone receptor-β gene in 164 Spanish and 2 Greek patients with resistance to thyroid hormone.

Authors:  Antonio J Amor; Irene Halperin; Rocίo Alfayate; Victoria M Borrás; Arancha Escribano; Cintia González; Antonio Gutirrez; Montserrat Mauri; Pilar Pérez; Antonio Picό; Irene Vourliotaki; Josep Oriola
Journal:  Hormones (Athens)       Date:  2014 Jan-Mar       Impact factor: 2.885

8.  Resistance to thyroid hormone due to a novel mutation of thyroid hormone receptor beta gene.

Authors:  Jae Hee Lee; Eun Young Kim
Journal:  Ann Pediatr Endocrinol Metab       Date:  2014-12-31

9.  A rare thyroid hormone receptor beta (THRβ) gene mutation in a 15-year-old girl with thyroid hormone resistance syndrome: a case report.

Authors:  Kawther El Shafie; Allal Ouhtit; Yousuf Al Farsi; Abeer Al Sayegh; Mohammed Al Shafaee
Journal:  J Med Case Rep       Date:  2014-01-06

10.  A newly identified insertion mutation in the thyroid hormone receptor-beta gene in a Korean family with generalized thyroid hormone resistance.

Authors:  Ji Hye Kim; Tae Sun Park; Hong Sun Baek; Gu Hwan Kim; Han Wook Yoo; Ji Hyun Park
Journal:  J Korean Med Sci       Date:  2007-06       Impact factor: 2.153

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  1 in total

1.  The Effect of High-Quality Nursing Management on Thyroid Tumor Patients after Bipolar Coagulation.

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Journal:  J Oncol       Date:  2022-03-19       Impact factor: 4.375

  1 in total

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