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Rare variants in genes encoding MuRF1 and MuRF2 are modifiers of hypertrophic cardiomyopathy
Journal article   Open access   Peer reviewed

Rare variants in genes encoding MuRF1 and MuRF2 are modifiers of hypertrophic cardiomyopathy

Ming Su, Jizheng Wang, Lianming Kang, Yilu Wang, Yubao Zou, Xinxing Feng, Dong Wang, Ferhaan Ahmad, Xianliang Zhou, Rutai Hui, …
International journal of molecular sciences, Vol.15(6), pp.9302-9313
05/26/2014
DOI: 10.3390/ijms15069302
PMCID: PMC4100095
PMID: 24865491
url
https://doi.org/10.3390/ijms15069302View
Published (Version of record) Open Access

Abstract

Modifier genes contribute to the diverse clinical manifestations of hypertrophic cardiomyopathy (HCM), but are still largely unknown. Muscle ring finger (MuRF) proteins are a class of muscle-specific ubiquitin E3-ligases that appear to modulate cardiac mass and function by regulating the ubiquitin-proteasome system. In this study we screened all the three members of the MuRF family, MuRF1, MuRF2 and MuRF3, in 594 unrelated HCM patients and 307 healthy controls by targeted resequencing. Identified rare variants were confirmed by capillary Sanger sequencing. The prevalence of rare variants in both MuRF1 and MuRF2 in HCM patients was higher than that in control subjects (MuRF1 13/594 (2.2%) vs. 1/307 (0.3%), p = 0.04; MuRF2 22/594 (3.7%) vs. 2/307 (0.7%); p = 0.007). Patients with rare variants in MuRF1 or MuRF2 were younger (p = 0.04) and had greater maximum left ventricular wall thickness (p = 0.006) than those without such variants. Mutations in genes encoding sarcomere proteins were present in 19 (55.9%) of the 34 HCM patients with rare variants in MuRF1 and MuRF2. These data strongly supported that rare variants in MuRF1 and MuRF2 are associated with higher penetrance and more severe clinical manifestations of HCM. The findings suggest that dysregulation of the ubiquitin-proteasome system contributes to the pathogenesis of HCM.
Mutation Cardiomyopathy, Hypertrophic - genetics Humans Middle Aged Male Myocardium - pathology Tripartite Motif Proteins Genetic Variation Muscle Proteins - genetics Myocardium - metabolism Adult Female Ubiquitin-Protein Ligases - genetics Cardiomyopathy, Hypertrophic - pathology

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