Literature DB >> 9541102

Genetic heterogeneity and HOMOG analysis in British malignant hyperthermia families.

R Robinson1, J L Curran, W J Hall, P J Halsall, P M Hopkins, A F Markham, A D Stewart, S P West, F R Ellis.   

Abstract

Malignant hyperthermia (MH) is an autosomal dominant genetic condition that presents in susceptible people undergoing general anaesthesia. The clinical disorder is a major cause of anaesthetic morbidity and mortality. The UK Malignant Hyperthermia Group has performed genetic linkage analysis on 20 large, well defined malignant hyperthermia families, using hypervariable markers on chromosome 19q13.1, including the candidate MH gene RYR1, the gene coding for the skeletal muscle ryanodine receptor protein. The results were analysed using LINKAGE to perform two point and multipoint lod scores, then HOMOG to calculate levels of heterogeneity. The results clearly showed genetic heterogeneity between MH families; nine of the families gave results entirely consistent with linkage to the region around RYR1 while the same region was clearly excluded in three families. In the remaining eight MHS families there were single recombinant events between RYR1 and MH susceptibility. HOMOG analysis was of little added benefit in determining the likelihood of linkage to RYR1 in these families. This confirmation of the presence of heterogeneity in the UK MH population, along with the possibility of the presence of two MH genes in some pedigrees, indicates that it would be premature and potentially dangerous to offer diagnosis of MH by DNA based methods at this time.

Entities:  

Mesh:

Substances:

Year:  1998        PMID: 9541102      PMCID: PMC1051241          DOI: 10.1136/jmg.35.3.196

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  25 in total

1.  A second-generation linkage map of the human genome.

Authors:  J Weissenbach; G Gyapay; C Dib; A Vignal; J Morissette; P Millasseau; G Vaysseix; M Lathrop
Journal:  Nature       Date:  1992-10-29       Impact factor: 49.962

2.  Dinucleotide repeat polymorphism at the RYR1 locus (19q13.1).

Authors:  F J Couch; K Hogan; T V McCarthy; R G Gregg
Journal:  Nucleic Acids Res       Date:  1991-09-25       Impact factor: 16.971

3.  Evidence for genetic heterogeneity of malignant hyperthermia susceptibility.

Authors:  T Deufel; A Golla; D Iles; A Meindl; T Meitinger; D Schindelhauer; A DeVries; D Pongratz; D H MacLennan; K J Johnson
Journal:  Am J Hum Genet       Date:  1992-06       Impact factor: 11.025

4.  The G1021A substitution in the RYR1 gene does not cosegregate with malignant hyperthermia susceptibility in a British pedigree.

Authors:  A M Adeokun; S P West; F R Ellis; P J Halsall; P M Hopkins; A M Foroughmand; D E Iles; R L Robinson; A D Stewart; J L Curran
Journal:  Am J Hum Genet       Date:  1997-04       Impact factor: 11.025

5.  A genome wide search for susceptibility loci in three European malignant hyperthermia pedigrees.

Authors:  R L Robinson; N Monnier; W Wolz; M Jung; A Reis; G Nuernberg; J L Curran; K Monsieurs; P Stieglitz; L Heytens; R Fricker; C van Broeckhoven; T Deufel; P M Hopkins; J Lunardi; C R Mueller
Journal:  Hum Mol Genet       Date:  1997-06       Impact factor: 6.150

6.  The structural organization of the human skeletal muscle ryanodine receptor (RYR1) gene.

Authors:  M S Phillips; J Fujii; V K Khanna; S DeLeon; K Yokobata; P J de Jong; D H MacLennan
Journal:  Genomics       Date:  1996-05-15       Impact factor: 5.736

7.  Genetic mapping of the beta 1- and gamma-subunits of the human skeletal muscle L-type voltage-dependent calcium channel on chromosome 17q and exclusion as candidate genes for malignant hyperthermia susceptibility.

Authors:  D E Iles; B Segers; R C Sengers; K Monsieurs; L Heytens; P J Halsall; P M Hopkins; F R Ellis; J L Hall-Curran; A D Stewart
Journal:  Hum Mol Genet       Date:  1993-07       Impact factor: 6.150

8.  Evidence for genetic heterogeneity in malignant hyperthermia susceptibility.

Authors:  R C Levitt; N Nouri; A E Jedlicka; V A McKusick; A R Marks; J G Shutack; J E Fletcher; H Rosenberg; D A Meyers
Journal:  Genomics       Date:  1991-11       Impact factor: 5.736

9.  Discordance, in a malignant hyperthermia pedigree, between in vitro contracture-test phenotypes and haplotypes for the MHS1 region on chromosome 19q12-13.2, comprising the C1840T transition in the RYR1 gene.

Authors:  T Deufel; R Sudbrak; Y Feist; B Rübsam; I Du Chesne; K L Schäfer; N Roewer; T Grimm; F Lehmann-Horn; E J Hartung
Journal:  Am J Hum Genet       Date:  1995-06       Impact factor: 11.025

10.  The work of the Leeds Malignant Hyperpyrexia Unit, 1971-84.

Authors:  F R Ellis; P J Halsall; D G Harriman
Journal:  Anaesthesia       Date:  1986-08       Impact factor: 6.955

View more
  3 in total

1.  Sudden death due to malignant hyperthermia with a mutation of RYR1: autopsy, morphology and genetic analysis.

Authors:  Wenhe Li; Lin Zhang; Yue Liang; Fang Tong; Yiwu Zhou
Journal:  Forensic Sci Med Pathol       Date:  2017-11-04       Impact factor: 2.007

2.  Ankyrin-B is required for intracellular sorting of structurally diverse Ca2+ homeostasis proteins.

Authors:  S Tuvia; M Buhusi; L Davis; M Reedy; V Bennett
Journal:  J Cell Biol       Date:  1999-11-29       Impact factor: 10.539

3.  Functional analysis of RYR1 variants linked to malignant hyperthermia.

Authors:  Jeremy Stephens; Anja H Schiemann; Cornelia Roesl; Dorota Miller; Sean Massey; Neil Pollock; Terasa Bulger; Kathryn Stowell
Journal:  Temperature (Austin)       Date:  2016-02-26
  3 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.