Literature DB >> 2493313

[Research on genetic abnormality in the hemolytic form of hereditary elliptocytosis with homozygosity for the spectrin alpha I/74 variant].

M Garbarz1, I Devaux, B Grandchamp, C Picat, D Dhermy, M C Lecomte, P Boivin, K E Sahr, B Forget.   

Abstract

We have undertaken to identify the spectrin gene mutation in a patient with a severe hemolytic form of Hereditary Elliptocytosis with homozygosity for the spectrin alpha I/74 variant. This variant corresponds to the presence of a 74,000 peptide which is produced during mild tryptic digestion of spectrin by cleavage at the Arginine-39 of the alpha I/80,000 domain of the spectrin alpha chain (595 amino acids). We hypothesized that the alpha I/74 mutation would be closed to the cleavage site Arg-39. A genomic library built with the patient's DNA was screened with a probe corresponding to a fragment of the alpha spectrin gene. Two clones were isolated, one being of paternal, the other of maternal origin. The subclones obtained contained the alpha spectrin gene exons 2 and 3 which encode for the first 88 amino-acids of the spectrin alpha I domain. The sequences obtained did not show any abnormality. The implications of these results are discussed.

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Year:  1989        PMID: 2493313

Source DB:  PubMed          Journal:  C R Acad Sci III        ISSN: 0764-4469


  3 in total

1.  Two elliptocytogenic alpha I/74 variants of the spectrin alpha I domain. Spectrin Culoz (GGT----GTT; alpha I 40 Gly----Val) and spectrin Lyon (CTT----TTT; alpha I 43 Leu---Phe).

Authors:  L Morlé; A F Roux; N Alloisio; B Pothier; J Starck; L Denoroy; F Morlé; R C Rudigoz; B G Forget; J Delaunay
Journal:  J Clin Invest       Date:  1990-08       Impact factor: 14.808

2.  Point mutation in the beta-spectrin gene associated with alpha I/74 hereditary elliptocytosis. Implications for the mechanism of spectrin dimer self-association.

Authors:  W T Tse; M C Lecomte; F F Costa; M Garbarz; C Feo; P Boivin; D Dhermy; B G Forget
Journal:  J Clin Invest       Date:  1990-09       Impact factor: 14.808

3.  Sequence and exon-intron organization of the DNA encoding the alpha I domain of human spectrin. Application to the study of mutations causing hereditary elliptocytosis.

Authors:  K E Sahr; T Tobe; A Scarpa; K Laughinghouse; S L Marchesi; P Agre; A J Linnenbach; V T Marchesi; B G Forget
Journal:  J Clin Invest       Date:  1989-10       Impact factor: 14.808

  3 in total

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