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PMID: 3684597 Published · ppublish English Case Reports Comparative Study Journal Article Research Support, Non-U.S. Gov't

Severe combined immune deficiency due to a homozygous 3.2-kb deletion spanning the promoter and first exon of the adenosine deaminase gene.

Nucleic acids research ·Vol. 15 ·No. 22 ·1987-11-25 ·Pages 9365-78

Berkvens TM, Gerritsen EJ, Oldenburg M, Breukel C, Wijnen JT, van Ormondt H, Vossen JM, van der Eb AJ, Meera Khan P

Abstract

We have investigated the structural gene for adenosine deaminase (ADA) in a female infant with ADA deficiency associated severe combined immune deficiency (ADA-SCID) disease and her family by DNA restriction-fragment-length analysis. In this family a new ADA-specific restriction-fragment-length variant was detected, which involves a 3.2-kb deletion spanning the ADA promoter as well as the first exon. It was found that the patient, who was born to a consanguineous couple, was homozygous and both her parents and her brother were heterozygous for the deletion. No ADA-specific mRNA could be detected by hybridization in fibroblasts derived from this patient. Thus the patient was established to be homozygous for a true null ADA allele. In the light of the apparently normal development of most tissues except the lymphoid tissue the above finding directly questions the classification of ADA as a 'housekeeping' enzyme.

MeSH Terms
Adenosine Deaminase/blood,deficiency Chromosome Deletion Erythrocytes/enzymology Exons Female Genes Homozygote Humans Immunologic Deficiency Syndromes/enzymology,genetics Infant Leukocytes/enzymology Male Nucleoside Deaminases/deficiency Promoter Regions, Genetic RNA, Messenger/genetics Reference Values Transcription, Genetic
Chemicals
RNA, Messenger Nucleoside Deaminases Adenosine Deaminase
Authors & Affiliations
9 authors, click to expand affiliations / ORCID
Berkvens T M
Department of Human Genetics, Sylvius Laboratories, University of Leiden, The Netherlands.
Gerritsen E J
Oldenburg M
Breukel C
Wijnen J T
van Ormondt H
Vossen J M
van der Eb A J
Meera Khan P
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Article Info
Journal
Nucleic acids research
Abbr.
Nucleic Acids Res
ISSN
0305-1048
Published
1987-11-25
Pages
9365-78
Language
English
Region
England
NLM ID
0411011
PMCID
PMC306474
Subset
IM
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