Literature DB >> 25450489

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

Michelle S Parvatiyar1, Jose Renato Pinto2.   

Abstract

BACKGROUND: Dilated and hypertrophic cardiomyopathy mutations in troponin can blunt effects of protein kinase A (PKA) phosphorylation of cardiac troponin I (cTnI), decreasing myofilament Ca2+-sensitivity; however this effect has never been tested for restrictive cardiomyopathy (RCM) mutants. This study explores whether an RCM cardiac troponin T mutant (cTnT-ΔE96) interferes with convergent PKA regulation and if TnT instability contributes to greatly enhanced Ca2+-sensitivity in skinned fibers.
METHODS: Force of contraction in skinned cardiac porcine fiber and spectroscopic studies were performed.
RESULTS: A decrease of -0.26 and -0.25 pCa units in Ca2+-sensitivity of contraction after PKA incubation was observed for skinned fibers incorporated with WT or cTnT-ΔE96, respectively. To further assess whether cTnT-ΔE96 interferes solely with transmission of cTnI phosphorylation effects, skinned fibers were reconstituted with PKA pseudo-phosphorylated cTnI (cTnI-SS/DD.cTnC). Fibers displaced with cTnT-WT, reconstituted with cTnI-SS/DD.cTnC decreased Ca2+-sensitivity of force (pCa50=5.61) compared to control cTnI-WT.cTnC (pCa50=5.75), similarly affecting cTnT-ΔE96 (pCa50=6.03) compared to control \cTnI-WT.cTnC (pCa50=6.14). Fluorescence studies measuring cTnC(IAANS) Ca2+-affinity changes due to cTnT-ΔE96 indicated that higher complexity (thin filament) better recapitulates skinned fiber Ca2+ sensitive changes. Circular dichroism revealed reduced α-helicity and earlier thermal unfolding for cTnT-ΔE96 compared to WT.
CONCLUSIONS: Although ineffective in decreasing myofilament Ca2+-sensitivity to normal levels, cTnT-ΔE96 does not interfere with PKA cTnI phosphorylation mediated effects; 2) cTnT-ΔE96 requires actin to increase cTnC Ca2+-affinity; and 3) deletion of E96 reduces cTnT stability, likely disrupting crucial thin filament interactions. GENERAL SIGNIFICANCE: The pathological effect of cTnT-ΔE96 is largely manifested by dramatic myofilament Ca2+-sensitization which still persists even after PKA phosphorylation mediated Ca2+-desensitization.
Copyright © 2014 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Cardiac troponin T; Circular dichroism; Fluorescence; Restrictive cardiomyopathy; Skinned fiber; Troponin I phosphorylation

Mesh:

Substances:

Year:  2014        PMID: 25450489      PMCID: PMC4276470          DOI: 10.1016/j.bbagen.2014.09.029

Source DB:  PubMed          Journal:  Biochim Biophys Acta        ISSN: 0006-3002


  61 in total

1.  Conserved Asp-137 imparts flexibility to tropomyosin and affects function.

Authors:  John P Sumida; Eleanor Wu; Sherwin S Lehrer
Journal:  J Biol Chem       Date:  2007-12-29       Impact factor: 5.157

Review 2.  Multiplex kinase signaling modifies cardiac function at the level of sarcomeric proteins.

Authors:  R John Solaro
Journal:  J Biol Chem       Date:  2008-06-19       Impact factor: 5.157

Review 3.  Thin filament mutations: developing an integrative approach to a complex disorder.

Authors:  Jil C Tardiff
Journal:  Circ Res       Date:  2011-03-18       Impact factor: 17.367

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

Authors:  Shi Wei Yang; Marc-Phillip Hitz; Gregor Andelfinger
Journal:  Cardiol Young       Date:  2010-06-23       Impact factor: 1.093

5.  Investigation of a transgenic mouse model of familial dilated cardiomyopathy.

Authors:  Weihua Song; Emma Dyer; Daniel Stuckey; Man-Ching Leung; Massimiliano Memo; Catherine Mansfield; Michael Ferenczi; Ke Liu; Charles Redwood; Kristen Nowak; Sian Harding; Kieran Clarke; Dominic Wells; Steven Marston
Journal:  J Mol Cell Cardiol       Date:  2010-06-17       Impact factor: 5.000

6.  Challenging current paradigms related to cardiomyopathies. Are changes in the Ca2+ sensitivity of myofilaments containing cardiac troponin C mutations (G159D and L29Q) good predictors of the phenotypic outcomes?

Authors:  David Dweck; Nir Hus; James D Potter
Journal:  J Biol Chem       Date:  2008-09-26       Impact factor: 5.157

Review 7.  Cardiac troponin mutations and restrictive cardiomyopathy.

Authors:  Michelle S Parvatiyar; Jose Renato Pinto; David Dweck; James D Potter
Journal:  J Biomed Biotechnol       Date:  2010-06-08

8.  A functional and structural study of troponin C mutations related to hypertrophic cardiomyopathy.

Authors:  Jose Renato Pinto; Michelle S Parvatiyar; Michelle A Jones; Jingsheng Liang; Michael J Ackerman; James D Potter
Journal:  J Biol Chem       Date:  2009-05-12       Impact factor: 5.157

9.  Idiopathic restrictive cardiomyopathy in children is caused by mutations in cardiac sarcomere protein genes.

Authors:  J P Kaski; P Syrris; M Burch; M-T Tomé-Esteban; M Fenton; M Christiansen; P S Andersen; N Sebire; M Ashworth; J E Deanfield; W J McKenna; P M Elliott
Journal:  Heart       Date:  2008-05-08       Impact factor: 5.994

Review 10.  Mutations in Troponin that cause HCM, DCM AND RCM: what can we learn about thin filament function?

Authors:  Ruth H Willott; Aldrin V Gomes; Audrey N Chang; Michelle S Parvatiyar; Jose Renato Pinto; James D Potter
Journal:  J Mol Cell Cardiol       Date:  2009-11-12       Impact factor: 5.000

View more
  2 in total

1.  Overexpression of miRNA-9 Generates Muscle Hypercontraction Through Translational Repression of Troponin-T in Drosophila melanogaster Indirect Flight Muscles.

Authors:  Prasanna Katti; Divesh Thimmaya; Aditi Madan; Upendra Nongthomba
Journal:  G3 (Bethesda)       Date:  2017-10-05       Impact factor: 3.154

2.  Variant R94C in TNNT2-Encoded Troponin T Predisposes to Pediatric Restrictive Cardiomyopathy and Sudden Death Through Impaired Thin Filament Relaxation Resulting in Myocardial Diastolic Dysfunction.

Authors:  Jordan E Ezekian; Sarah R Clippinger; Jaquelin M Garcia; Qixin Yang; Susan Denfield; Aamir Jeewa; William J Dreyer; Wenxin Zou; Yuxin Fan; Hugh D Allen; Jeffrey J Kim; Michael J Greenberg; Andrew P Landstrom
Journal:  J Am Heart Assoc       Date:  2020-02-26       Impact factor: 5.501

  2 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.