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PMID: 9329953 Published · ppublish English Comparative Study Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

A p47-phox pseudogene carries the most common mutation causing p47-phox- deficient chronic granulomatous disease.

The Journal of clinical investigation ·Vol. 100 ·No. 8 ·1997-10-15 ·Pages 1907-18

Görlach A, Lee PL, Roesler J, Hopkins PJ, Christensen B, Green ED, Chanock SJ, Curnutte JT

Abstract

The predominant genetic defect causing p47-phox-deficient chronic granulomatous disease (A47 degrees CGD) is a GT deletion (DeltaGT) at the beginning of exon 2. No explanation exists to account for the high incidence of this single mutation causing a rare disease in an unrelated, racially diverse population. In each of 34 consecutive unrelated normal individuals, both the normal and mutant DeltaGT sequences were present in genomic DNA, suggesting that a p47-phox related sequence carrying DeltaGT exists in the normal population. Screening of genomic bacteriophage and YAC libraries identified 13 p47-phox bacteriophage and 19 YAC clones. The GT deletion was found in 11 bacteriophage and 15 YAC clones. Only 5 exonic and 33 intronic differences distinguished all DeltaGT clones from all wild-type clones. The most striking differences were a 30-bp deletion in intron 1 and a 20-bp duplication in intron 2. These results provide good evidence for the existence of at least one highly homologous p47-phox pseudogene containing the DeltaGT mutation. The p47-phox gene and pseudogene(s) colocalize to chromosome 7q11.23. This close linkage, together with the presence within each gene of multiple recombination hot spots, suggests that the predominance of the DeltaGT mutation in A47 degrees CGD is caused by recombination events between the wild-type gene and the pseudogene(s).

MeSH Terms
Bacteriophages/genetics Chromosome Mapping Chromosomes, Artificial, Yeast/genetics Chromosomes, Human, Pair 7 Exons Gene Dosage Genetic Variation Genomic Library Granulomatous Disease, Chronic/etiology,genetics Humans Introns Mutation NADPH Dehydrogenase NADPH Oxidases Neutrophils/enzymology Phosphoproteins/deficiency,genetics Polymerase Chain Reaction Pseudogenes Recombination, Genetic Repetitive Sequences, Nucleic Acid Sequence Analysis, DNA Sequence Deletion
Chemicals
Phosphoproteins NADPH Oxidases neutrophil cytosolic factor 1 NADPH Dehydrogenase
Authors & Affiliations
8 authors, click to expand affiliations / ORCID
Görlach A
The Scripps Research Institute, Department of Molecular & Experimental Medicine, La Jolla, California 92037, USA.
Lee P L
Roesler J
Hopkins P J
Christensen B
Green E D
Chanock S J
Curnutte J T
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Article Info
Journal
The Journal of clinical investigation
Abbr.
J Clin Invest
ISSN
0021-9738
Published
1997-10-15
Pages
1907-18
Language
English
Region
United States
NLM ID
7802877
PMCID
PMC508379
Subset
IM
Grants
NIAID NIH HHS · R01 AI24838 · United States
NCRR NIH HHS · RR00833 · United States
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GENBANK
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