Literature DB >> 20569525

Ventricular septal defect and restrictive cardiomyopathy in a paediatric TNNI3 mutation carrier.

Shi Wei Yang1, Marc-Phillip Hitz, Gregor Andelfinger.   

Abstract

Here, we report an infantile case of ventricular septal defect and restrictive cardiomyopathy caused by a de novo mutation of the cardiac troponin T gene. Initially diagnosed with a perimembranous ventricular septal defect in the newborn period, we observed the first signs of restrictive cardiomyopathy at the age of 18 months. The patient was listed for cardiac transplantation at the age of 8 years despite optimal medical treatment. The finding of a de novo mutation in TNNI3 (R204H) enabled a genetic diagnosis and counselling. We suggest that the previously reported overlap of functional and morphologic phenotypes in sarcomeric genes may also be a feature of TNNI3 mutations.

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Year:  2010        PMID: 20569525     DOI: 10.1017/S1047951110000715

Source DB:  PubMed          Journal:  Cardiol Young        ISSN: 1047-9511            Impact factor:   1.093


  8 in total

1.  Pathogenesis associated with a restrictive cardiomyopathy mutant in cardiac troponin T is due to reduced protein stability and greatly increased myofilament Ca2+ sensitivity.

Authors:  Michelle S Parvatiyar; Jose Renato Pinto
Journal:  Biochim Biophys Acta       Date:  2014-11-01

Review 2.  Genetic Insights into Primary Restrictive Cardiomyopathy.

Authors:  Andreas Brodehl; Brenda Gerull
Journal:  J Clin Med       Date:  2022-04-08       Impact factor: 4.964

3.  Application of echocardiography on transgenic mice with cardiomyopathies.

Authors:  G Chen; Y Li; J Tian; L Zhang; P Jean-Charles; N Gobara; C Nan; J-P Jin; X P Huang
Journal:  Biochem Res Int       Date:  2012-05-21

4.  Novel GATA4 mutations in patients with congenital ventricular septal defects.

Authors:  Yi-Qing Yang; Juan Wang; Xing-Yuan Liu; Xiao-Zhong Chen; Wei Zhang; Xiao-Zhou Wang; Xu Liu; Wei-Yi Fang
Journal:  Med Sci Monit       Date:  2012-06

5.  Insights into restrictive cardiomyopathy from clinical and animal studies.

Authors:  Pierre-Yves Jean-Charles; Yue-Jin Li; Chang-Long Nan; Xu-Pei Huang
Journal:  J Geriatr Cardiol       Date:  2011-09       Impact factor: 3.327

6.  Troponin Mutation Caused Diastolic Dysfunction and Experimental Treatment in Transgenic Mice with Cardiomyopathy.

Authors:  Yang Xu; Jie Tian; Xupei Huang
Journal:  GSTF J Adv Med Res       Date:  2015-07-09

7.  Discovery of a potentially deleterious variant in TMEM87B in a patient with a hemizygous 2q13 microdeletion suggests a recessive condition characterized by congenital heart disease and restrictive cardiomyopathy.

Authors:  Hung-Chun Yu; Curtis R Coughlin; Elizabeth A Geiger; Blake J Salvador; Ellen R Elias; Jean L Cavanaugh; Kathryn C Chatfield; Shelley D Miyamoto; Tamim H Shaikh
Journal:  Cold Spring Harb Mol Case Stud       Date:  2016-05

Review 8.  Untying the knot: protein quality control in inherited cardiomyopathies.

Authors:  Larissa M Dorsch; Maike Schuldt; Dora Knežević; Marit Wiersma; Diederik W D Kuster; Jolanda van der Velden; Bianca J J M Brundel
Journal:  Pflugers Arch       Date:  2018-08-14       Impact factor: 3.657

  8 in total

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