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April 12, 2013Journal of Biological ChemistryOpen Access

Transgenic Mouse α- and β-Cardiac Myosins Containing the R403Q Mutation Show Isoform-dependent Transient Kinetic Differences

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Key result

The R403Q mutation enhanced the ADP release rate by 20% (to 430 s−1) in mouse α-cardiac myosin but caused only a minor reduction in kinetics for β-cardiac myosin.

Population

Transgenic mouse model in which endogenous α-MHC was partially replaced with transgenically encoded β-MHC or…

Comparison

R403Q mutation in α-S1 and β-S1 cardiac myosin… vs Wild-type α-S1 and β-S1 cardiac myosin isoforms

Design

Preclinical

Authors

SLSusan LoweyVBVera BrettonJGJames Gulick

Discussion

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Overview

Isoform-dependent R403Q effects caution against α-MHC mouse models for human β-MHC HCM; leaves open need for matched isoform studies.

Structured PICO

P
Population
Transgenic mouse model in which endogenous α-MHC was partially replaced with transgenically encoded β-MHC or α-MHC containing the R403Q mutation and a His6 tag
I
Intervention
R403Q mutation in α-S1 and β-S1 cardiac myosin isoforms
C
Comparator
Wild-type α-S1 and β-S1 cardiac myosin isoforms
O
Outcome
Equilibrium constants and rates of nucleotide binding and release (MgADP affinity and release rate) measured by stopped flow kineticssurrogate

The functional impact of the R403Q mutation on cardiac myosin kinetics depends on the isoform backbone, indicating that mouse models expressing α-MHC may not fully recapitulate the human hypertrophic cardiomyopathy phenotype driven by β-MHC.

Cite This Study

Lowey et al. (2013) studied Familial hypertrophic cardiomyopathy. R403Q mutation in α- and β-cardiac myosin heavy chain vs. Wild-type isoforms was evaluated on Equilibrium constants and rates of nucleotide binding and release. The R403Q mutation enhanced the ADP release rate by 20% (to 430 s−1) in mouse α-cardiac myosin but caused only a minor reduction in kinetics for β-cardiac myosin.

synapsesocial.com/papers/6a79c01e02ab7a73dd028901https://doi.org/10.1074/jbc.m113.450668
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Also Consider

Synapse has enriched 5 closely related papers on similar clinical questions. Consider them for comparative context:

  1. 1Functional Effects of the Hypertrophic Cardiomyopathy R403Q Mutation Are Different in an α- or β-Myosin Heavy Chain Backbone2008 · 93 citations
  2. 2Altered Crossbridge Kinetics in the αMHC <sup>403/+</sup> Mouse Model of Familial Hypertrophic Cardiomyopathy1999 · 101 citations
  3. 3Differential cross-bridge kinetics of FHC myosin mutations R403Q and R453C in heterozygous mouse myocardium2004 · 80 citations
  4. 4The R403Q Myosin Mutation Implicated in Familial Hypertrophic Cardiomyopathy Causes Disorder at the Actomyosin Interface2007 · 30 citations
  5. 5Hypertrophic cardiomyopathy R403Q mutation in rabbit β-myosin reduces contractile function at the molecular and myofibrillar levels2018 · 40 citations