Literature DB >> 8220423

Mutations in the ryanodine receptor gene in central core disease and malignant hyperthermia.

K A Quane1, J M Healy, K E Keating, B M Manning, F J Couch, L M Palmucci, C Doriguzzi, T H Fagerlund, K Berg, H Ording.   

Abstract

Central core disease (CCD) of muscle is an inherited myopathy which is closely associated with malignant hyperthermia (MH) in humans. CCD has recently been shown to be tightly linked to the ryanodine receptor gene (RYR1) and mutations in this gene are known to be present in MH. Mutation screening of RYR1 has led to the identification of two previously undescribed mutations in different CCD pedigrees. One of these mutations was also detected in an unrelated MH pedigree whose members are asymptomatic of CCD. The data suggest a model to explain how a single mutation may result in two apparently distinct clinical phenotypes.

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Year:  1993        PMID: 8220423     DOI: 10.1038/ng0993-51

Source DB:  PubMed          Journal:  Nat Genet        ISSN: 1061-4036            Impact factor:   38.330


  74 in total

Review 1.  Ion channels and neurology.

Authors:  S M Zuberi; M G Hanna
Journal:  Arch Dis Child       Date:  2001-03       Impact factor: 3.791

Review 2.  Ion channel genes and human neurological disease: recent progress, prospects, and challenges.

Authors:  E C Cooper; L Y Jan
Journal:  Proc Natl Acad Sci U S A       Date:  1999-04-27       Impact factor: 11.205

3.  Ion channels in health and disease. 83rd Boehringer Ingelheim Fonds International Titisee Conference.

Authors:  B A Niemeyer; L Mery; C Zawar; A Suckow; F Monje; L A Pardo; W Stuhmer; V Flockerzi; M Hoth
Journal:  EMBO Rep       Date:  2001-07       Impact factor: 8.807

Review 4.  Unraveling monogenic channelopathies and their implications for complex polygenic disease.

Authors:  J Jay Gargus
Journal:  Am J Hum Genet       Date:  2003-03-07       Impact factor: 11.025

5.  Excitation--contraction uncoupling by a human central core disease mutation in the ryanodine receptor.

Authors:  G Avila; J J O'Brien; R T Dirksen
Journal:  Proc Natl Acad Sci U S A       Date:  2001-03-27       Impact factor: 11.205

6.  Detection of a novel mutation in the ryanodine receptor gene in an Irish malignant hyperthermia pedigree: correlation of the IVCT response with the affected and unaffected haplotypes.

Authors:  K E Keating; L Giblin; P J Lynch; K A Quane; M Lehane; J J Heffron; T V McCarthy
Journal:  J Med Genet       Date:  1997-04       Impact factor: 6.318

7.  Central core disease mutations R4892W, I4897T and G4898E in the ryanodine receptor isoform 1 reduce the Ca2+ sensitivity and amplitude of Ca2+-dependent Ca2+ release.

Authors:  Guo Guang Du; Vijay K Khanna; Xinghua Guo; David H MacLennan
Journal:  Biochem J       Date:  2004-09-01       Impact factor: 3.857

8.  The pore structure of the closed RyR1 channel.

Authors:  Steven J Ludtke; Irina I Serysheva; Susan L Hamilton; Wah Chiu
Journal:  Structure       Date:  2005-08       Impact factor: 5.006

Review 9.  Congenital myopathies.

Authors:  Claudio Bruno; Carlo Minetti
Journal:  Curr Neurol Neurosci Rep       Date:  2004-01       Impact factor: 5.081

10.  A multi-dimensional analysis of genotype-phenotype discordance in malignant hyperthermia susceptibility.

Authors:  Carlos A Ibarra Moreno; Natalia Kraeva; Elena Zvaritch; Lourdes Figueroa; Eduardo Rios; Leslie Biesecker; Filip Van Petegem; Philip M Hopkins; Sheila Riazi
Journal:  Br J Anaesth       Date:  2020-08-27       Impact factor: 9.166

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