Literature DB >> 15813912

Prenatal diagnosis of recessive congenital methaemoglobinaemia type II: novel mutation in the NADH-cytochrome b5 reductase gene leading to stop codon read-through.

Alena Leroux1, France Leturcq, Nathalie Deburgrave, Marie-France Szajnert.   

Abstract

A case of type II recessive congenital methaemoglobinaemia (RCM) observed in a Lebanese subject with a novel mutation in NADH-cytochrome b5 reductase gene is described. A homozygous mutation CAC to AA identified at Thr 295 with an out-of-frame 1-bp deletion leads to a frameshift with translational read-through of the natural stop codon. The molecular mechanism is demonstrated by an in vitro translation study. The model of mutated cytochrome b5 reductase protein possessing 46 additional amino acids was obtained by homology modelling. The mutation causes an alteration of hydrophobicity in the carboxyl-terminal portion, resulting in the conformation being drastically disturbed by the presence of 46 supplementary amino acids. The identical mutation was found in the heterozygous state in the patient's parents and sister. Identification of this new mutation enabled us to perform the molecular prenatal diagnosis of type II RCM at the DNA level.

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Year:  2005        PMID: 15813912     DOI: 10.1111/j.1600-0609.2004.00388.x

Source DB:  PubMed          Journal:  Eur J Haematol        ISSN: 0902-4441            Impact factor:   2.997


  4 in total

1.  The severe autosomal dominant retinitis pigmentosa rhodopsin mutant Ter349Glu mislocalizes and induces rapid rod cell death.

Authors:  T J Hollingsworth; Alecia K Gross
Journal:  J Biol Chem       Date:  2013-08-12       Impact factor: 5.157

2.  A novel nine base deletion mutation in NADH-cytochrome b5 reductase gene in an Indian family with recessive congenital methemoglobinemia-type-II.

Authors:  Prashant Warang; Prabhakar Kedar; S Sivanandam; K Jothilakshmi; R Sumathi; Roshan Colah
Journal:  Mol Genet Metab Rep       Date:  2015-10-22

3.  Neurological and Neuroimaging Features of CYB5R3-Related Recessive Hereditary Methemoglobinemia Type II.

Authors:  Francesco Nicita; Letizia Sabatini; Viola Alesi; Giulia Lucignani; Ester Sallicandro; Antonella Sferra; Enrico Bertini; Ginevra Zanni; Giuseppe Palumbo
Journal:  Brain Sci       Date:  2022-01-29

4.  Congenital methemoglobinemia type II in a 5-year-old boy.

Authors:  Elizabeth A Mannino; Thomas Pluim; Jacob Wessler; Megan T Cho; Jane Juusola; Samantha A Schrier Vergano
Journal:  Clin Case Rep       Date:  2017-12-07
  4 in total

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