Literature DB >> 34076875

A Novel Mutation in the Myosin Binding Protein C Gene in a Prader-Willi Syndrome Pedigree.

Xiao-Qun Liu1,2, Man Luo1,2, Qi Liu3,4, Guo-Can Yang5,6.   

Abstract

Prader-Willi syndrome (PWS) is a neurogenetic disorder caused by deficiency expression of paternally imprinted genes of the chromosomal region 15. In this study, we report a novel mutation in the myosin binding protein C (MYBPC3) gene in a Prader-Willi syndrome pedigree. Next-generation sequencing (NGS) and Sanger sequencing were performed to define and confirm the MYBPC3 gene mutation. Bioinformatics analysis was also performed for the mutated MYBPC3 protein using available software tools. The proband was diagnosed as PWS with about 4.727Mb copy number missed in the long arm of chromosome 15 and treated with growth hormone on 0.3 IU/day. Sanger sequencing identified a novel heterozygous mutation in the MYBPC3 gene, c.2002C>G (p.R668G). Bioinformatics analysis suggested the variant disease-causing; the Pro residue at 668 in the MYBPC3 protein was highly conserved. Moreover, interactions among MYBPC3 and other proteins suggested the potential effects on the development of cardiomyopathies. This is the first report of PWS with MYBPC3 gene mutation. Besides general examinations, it is vital for physicians to amply molecular genetics to get an accurate diagnosis in the clinic especially for rare diseases.
© 2021. Society for Reproductive Investigation.

Entities:  

Keywords:  Hypertrophic cardiomyopathy; MYBPC3; Methylation; Prader-Willi syndrome; Rare diseases; p.R668G

Mesh:

Substances:

Year:  2021        PMID: 34076875     DOI: 10.1007/s43032-021-00620-4

Source DB:  PubMed          Journal:  Reprod Sci        ISSN: 1933-7191            Impact factor:   3.060


  13 in total

1.  Molecular genetic classification in Prader-Willi syndrome: a multisite cohort study.

Authors:  Merlin G Butler; Samantha N Hartin; Waheeda A Hossain; Ann M Manzardo; Virginia Kimonis; Elisabeth Dykens; June Anne Gold; Soo-Jeong Kim; Nicolette Weisensel; Roy Tamura; Jennifer L Miller; Daniel J Driscoll
Journal:  J Med Genet       Date:  2018-05-05       Impact factor: 6.318

2.  A Premature Termination Codon Mutation in MYBPC3 Causes Hypertrophic Cardiomyopathy via Chronic Activation of Nonsense-Mediated Decay.

Authors:  Timon Seeger; Rajani Shrestha; Chi Keung Lam; Caressa Chen; Wesley L McKeithan; Edward Lau; Alexa Wnorowski; George McMullen; Matthew Greenhaw; Jaecheol Lee; Angelos Oikonomopoulos; Soah Lee; Huaxiao Yang; Mark Mercola; Matthew Wheeler; Euan A Ashley; Fan Yang; Ioannis Karakikes; Joseph C Wu
Journal:  Circulation       Date:  2019-02-05       Impact factor: 29.690

3.  Three siblings with Prader-Willi syndrome caused by imprinting center microdeletions and review.

Authors:  Samantha N Hartin; Waheeda A Hossain; Nicolette Weisensel; Merlin G Butler
Journal:  Am J Med Genet A       Date:  2018-02-13       Impact factor: 2.802

4.  The changing purpose of Prader-Willi syndrome clinical diagnostic criteria and proposed revised criteria.

Authors:  M Gunay-Aygun; S Schwartz; S Heeger; M A O'Riordan; S B Cassidy
Journal:  Pediatrics       Date:  2001-11       Impact factor: 7.124

5.  Genotypic effect of a mutation of the MYBPC3 gene and two phenotypes with different patterns of inheritance.

Authors:  Nejat Mahdieh; Maryam Hosseini Moghaddam; Mahsa Motavaf; Ahmad Rabbani; Mahdieh Soveizi; Majid Maleki; Bahareh Rabbani; Azin Alizadeh-Asl
Journal:  J Clin Lab Anal       Date:  2018-03-01       Impact factor: 2.352

Review 6.  Consensus statement: chromosomal microarray is a first-tier clinical diagnostic test for individuals with developmental disabilities or congenital anomalies.

Authors:  David T Miller; Margaret P Adam; Swaroop Aradhya; Leslie G Biesecker; Arthur R Brothman; Nigel P Carter; Deanna M Church; John A Crolla; Evan E Eichler; Charles J Epstein; W Andrew Faucett; Lars Feuk; Jan M Friedman; Ada Hamosh; Laird Jackson; Erin B Kaminsky; Klaas Kok; Ian D Krantz; Robert M Kuhn; Charles Lee; James M Ostell; Carla Rosenberg; Stephen W Scherer; Nancy B Spinner; Dimitri J Stavropoulos; James H Tepperberg; Erik C Thorland; Joris R Vermeesch; Darrel J Waggoner; Michael S Watson; Christa Lese Martin; David H Ledbetter
Journal:  Am J Hum Genet       Date:  2010-05-14       Impact factor: 11.025

Review 7.  The dilemma of diagnostic testing for Prader-Willi syndrome.

Authors:  Arabella Smith; Dorothy Hung
Journal:  Transl Pediatr       Date:  2017-01

Review 8.  Prader-Willi Syndrome: Clinical Genetics and Diagnostic Aspects with Treatment Approaches.

Authors:  Merlin G Butler; Ann M Manzardo; Janice L Forster
Journal:  Curr Pediatr Rev       Date:  2016

9.  Prader-Willi syndrome: consensus diagnostic criteria.

Authors:  V A Holm; S B Cassidy; M G Butler; J M Hanchett; L R Greenswag; B Y Whitman; F Greenberg
Journal:  Pediatrics       Date:  1993-02       Impact factor: 7.124

10.  Analysis of the Prader-Willi syndrome imprinting center using droplet digital PCR and next-generation whole-exome sequencing.

Authors:  Samantha N Hartin; Waheeda A Hossain; David Francis; David E Godler; Sangjucta Barkataki; Merlin G Butler
Journal:  Mol Genet Genomic Med       Date:  2019-02-21       Impact factor: 2.183

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  1 in total

Review 1.  Proteins and proteases of Prader-Willi syndrome: a comprehensive review and perspectives.

Authors:  Sanjukta Basak; Ajoy Basak
Journal:  Biosci Rep       Date:  2022-06-30       Impact factor: 3.976

  1 in total

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