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PMID: 3358422 Published · ppublish English Case Reports Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

Localization of the McLeod locus (XK) within Xp21 by deletion analysis.

American journal of human genetics ·Vol. 42 ·No. 5 ·1988-05-00 ·Pages 703-11

Bertelson CJ, Pogo AO, Chaudhuri A, Marsh WL, Redman CM, Banerjee D, Symmans WA, Simon T, Frey D, Kunkel LM

Abstract

The McLeod phenotype is an X-linked, recessive disorder in which the red blood cells demonstrate acanthocytic morphology and weakened antigenicity in the Kell blood group system. The phenotype is associated with a reduction of in vivo red cell survival, but the permanent hemolytic state is usually compensated by erythropoietic hyperplasia. The McLeod phenotype is accompanied by either a subclinical myopathy and elevated creatine kinase (CK) or X-linked chronic granulomatous disease (CGD). Seven males with the McLeod red-blood-cell phenotype and associated myopathy but not CGD, one male with the McLeod phenotype associated with CGD, and two males known to possess large deletions of the Duchenne muscular dystrophy (DMD) locus were studied. DNA isolated from each patient was screened for the presence or absence of various cloned sequences located in the Xp21 region of the human X chromosome. Two of the seven males who have only the McLeod phenotype and are cousins exhibit deletions for four Xp21 cloned fragments but are not deleted for any portion of either the CGD or the DMD loci. Comparison of the cloned segments absent from these two McLeod cousins with those absent from the two DMD boys and the CGD/McLeod patient leads to the submapping of various cloned DNA segments within the Xp21 region. The results place the locus for the McLeod phenotype within a 500-kb interval distal from the CGD locus toward the DMD locus.

MeSH Terms
Adult Child Chromosome Deletion Cloning, Molecular DNA/genetics Granulomatous Disease, Chronic/genetics Humans Male Middle Aged Muscular Diseases/genetics Muscular Dystrophies/genetics Nucleic Acid Hybridization Phenotype Sex Chromosome Aberrations X Chromosome
Chemicals
DNA
Authors & Affiliations
10 authors, click to expand affiliations / ORCID
Bertelson C J
Howard Hughes Medical Institute, Boston, MA 02115.
Pogo A O
Chaudhuri A
Marsh W L
Redman C M
Banerjee D
Symmans W A
Simon T
Frey D
Kunkel L M
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Article Info
Journal
American journal of human genetics
Abbr.
Am J Hum Genet
ISSN
0002-9297
Published
1988-05-00
Pages
703-11
Language
English
Region
United States
NLM ID
0370475
PMCID
PMC1715185
Subset
IM
Grants
NICHD NIH HHS · HD18658 · United States
NHLBI NIH HHS · HL33841 · United States
NINDS NIH HHS · NS23740 · United States
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