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Wiskott-Aldrich syndrome carrier detection with the hypervariable marker M27 beta.

Authors :
de Saint Basile G
Notarangelo LD
Bonaiti-PelliƩ C
Doussau M
Prolini O
Craig IW
Ugazio A
Griscelli C
Fischer A
Source :
Human genetics [Hum Genet] 1992 May; Vol. 89 (2), pp. 223-8.
Publication Year :
1992

Abstract

Whole-blood cells of obligate carriers of the X-linked Wiskott-Aldrich syndrome (WAS) exhibit nonrandom inactivation of the X-chromosomes. However, because of the limited polymorphism of the probes available, the X-methylation pattern can only be determined in a restricted proportion of females. We thus analysed a large set of normal females and members of WAS families, using the recently described marker M27 beta, which detects the hyperpolymorphic locus DXS255. The probe was used to detect differences in methylation between the active and inactive X-chromosome, and the findings were compared with the pattern obtained using the well-documented probes from the 5' end of the PGK and HPRT genes. All the normal females were found to use either X-chromosome randomly, and there was complete correlation between the three probes in the populations studied. Segregation analysis performed with M27 beta and other related markers in the WAS families was fully in accordance with the X-inactivation data. The use of M27 beta, for both X-inactivation and segregation analysis of WAS kindreds, provides a basis for genetic counselling in the majority of families, including those with no surviving males.

Details

Language :
English
ISSN :
0340-6717
Volume :
89
Issue :
2
Database :
MEDLINE
Journal :
Human genetics
Publication Type :
Academic Journal
Accession number :
1350264
Full Text :
https://doi.org/10.1007/BF00217127