Literature DB >> 3752066

Sp alpha I/65 hereditary elliptocytosis in North Africa.

N Alloisio, D Guetarni, L Morlé, B Pothier, M T Ducluzeau, A Soun, P Colonna, M Clerc, N Philippe, J Delaunay.   

Abstract

The Sp alpha I/65 variant of the spectrin has been recently described in black people with hereditary elliptocytosis (HE). The present study reports on a similar Sp alpha I/65 variant in nine North African persons belonging to four unrelated families. The abnormality was associated with a variable degree of elliptocytosis. In one case, red cell morphology was normal. In the nine carriers of the biochemical abnormality, the spectrin dimer self-association was defective. The association constant was reduced: 0.65 to 1.7 X 10(5) M-1 (controls: 4.6 +/- 0.5 X 10(5) mM-1 (n = 21)); in six cases, there was a higher level of spectrin dimer in the low ionic strength extract at 4 degrees C: 13.0 to 19.7% (controls: 6.4 +/- 2.1% (n = 7)). Limited tryptic digests of spectrin from the nine persons revealed a decrease of the 80,000-dalton alpha-1 domain, and the concomitant appearance of a peptide with a molecular weight of 65,000 daltons and an isoelectric point ranging from 5.0 to 5.1. There was a correlation between the proportion of the 65,000-dalton fragments, the defect of spectrin self-association, and the extent of morphological alteration. This is the first large series concerning a spectrin abnormality in non-black persons. In North Africa, cases of HE that are not due to a protein 4.1 defect have turned out so far to be associated with the Sp alpha I/65 variant.

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Year:  1986        PMID: 3752066     DOI: 10.1002/ajh.2830230205

Source DB:  PubMed          Journal:  Am J Hematol        ISSN: 0361-8609            Impact factor:   10.047


  8 in total

1.  Alpha I/65 hereditary elliptocytosis in southern Italy: evidence for an African origin.

Authors:  E M del Giudice; M T Ducluzeau; N Alloisio; R Wilmotte; J Delaunay; S Perrotta; S Cutillo; A Iolascon
Journal:  Hum Genet       Date:  1992-07       Impact factor: 4.132

2.  Evidence that expression of Sp alpha I/65 hereditary elliptocytosis is compounded by a genetic factor that is linked to the homologous alpha-spectrin allele.

Authors:  D Guetarni; A F Roux; N Alloisio; F Morlé; M T Ducluzeau; B G Forget; P Colonna; J Delaunay; J Godet
Journal:  Hum Genet       Date:  1990-10       Impact factor: 4.132

3.  Sp alpha V/41: a common spectrin polymorphism at the alpha IV-alpha V domain junction. Relevance to the expression level of hereditary elliptocytosis due to alpha-spectrin variants located in trans.

Authors:  N Alloisio; L Morlé; J Maréchal; A F Roux; M T Ducluzeau; D Guetarni; B Pothier; F Baklouti; A Ghanem; R Kastally; J Delaunay
Journal:  J Clin Invest       Date:  1991-06       Impact factor: 14.808

4.  Low expression allele alpha LELY of red cell spectrin is associated with mutations in exon 40 (alpha V/41 polymorphism) and intron 45 and with partial skipping of exon 46.

Authors:  R Wilmotte; J Maréchal; L Morlé; F Baklouti; N Philippe; R Kastally; L Kotula; J Delaunay; N Alloisio
Journal:  J Clin Invest       Date:  1993-05       Impact factor: 14.808

5.  SP alpha I/65 hereditary elliptocytosis in Calabria (southern Italy).

Authors:  A Qualtieri; M G Bisconte; A Pasqua; M Bria; C Brancati
Journal:  Hum Genet       Date:  1995-03       Impact factor: 4.132

6.  Abnormal electrophoretic mobility of spectrin tetramers in hereditary elliptocytosis.

Authors:  D Dhermy; M Garbarz; M C Lecomte; I Chaveroche; O Bournier; H Gautero; I Blot; P Boivin
Journal:  Hum Genet       Date:  1986-12       Impact factor: 4.132

7.  Abnormal oxidant sensitivity and beta-chain structure of spectrin in hereditary spherocytosis associated with defective spectrin-protein 4.1 binding.

Authors:  P S Becker; J S Morrow; S E Lux
Journal:  J Clin Invest       Date:  1987-08       Impact factor: 14.808

8.  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

  8 in total

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