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PMID: 9463314 Published · ppublish English Case Reports Journal Article Research Support, Non-U.S. Gov't

ATM mutations and phenotypes in ataxia-telangiectasia families in the British Isles: expression of mutant ATM and the risk of leukemia, lymphoma, and breast cancer.

American journal of human genetics ·Vol. 62 ·No. 2 ·1998-02-00 ·Pages 334-45

Stankovic T, Kidd AM, Sutcliffe A, McGuire GM, Robinson P, Weber P, Bedenham T, Bradwell AR, Easton DF, Lennox GG, Haites N, Byrd PJ, Taylor AM

Abstract

We report the spectrum of 59 ATM mutations observed in ataxia-telangiectasia (A-T) patients in the British Isles. Of 51 ATM mutations identified in families native to the British Isles, 11 were founder mutations, and 2 of these 11 conferred a milder clinical phenotype with respect to both cerebellar degeneration and cellular features. We report, in two A-T families, an ATM mutation (7271T-->G) that may be associated with an increased risk of breast cancer in both homozygotes and heterozygotes (relative risk 12.7; P=. 0025), although there is a less severe A-T phenotype in terms of the degree of cerebellar degeneration. This mutation (7271T-->G) also allows expression of full-length ATM protein at a level comparable with that in unaffected individuals. In addition, we have studied 18 A-T patients, in 15 families, who developed leukemia, lymphoma, preleukemic T-cell proliferation, or Hodgkin lymphoma, mostly in childhood. A wide variety of ATM mutation types, including missense mutations and in-frame deletions, were seen in these patients. We also show that 25% of all A-T patients carried in-frame deletions or missense mutations, many of which were also associated with expression of mutant ATM protein.

MeSH Terms
Adolescent Adult Aged Aged, 80 and over Ataxia Telangiectasia/genetics Ataxia Telangiectasia Mutated Proteins Breast Neoplasms/epidemiology,genetics Cell Cycle Proteins Chromosome Mapping DNA-Binding Proteins Female Genetic Carrier Screening Genetic Markers Haplotypes Homozygote Humans Leucine Zippers Leukemia/epidemiology,genetics Lymphoma/epidemiology,genetics Male Middle Aged Mutation Pedigree Phenotype Point Mutation Protein Serine-Threonine Kinases Proteins/genetics Risk Factors Sequence Deletion Tumor Suppressor Proteins United Kingdom
Chemicals
Cell Cycle Proteins DNA-Binding Proteins Genetic Markers Proteins Tumor Suppressor Proteins ATM protein, human Ataxia Telangiectasia Mutated Proteins Protein Serine-Threonine Kinases
Authors & Affiliations
13 authors, click to expand affiliations / ORCID
Stankovic T
CRC Institute for Cancer Studies, The Medical School, University of Birmingham, United Kingdom.
Kidd A M
Sutcliffe A
McGuire G M
Robinson P
Weber P
Bedenham T
Bradwell A R
Easton D F
Lennox G G
Haites N
Byrd P J
Taylor A M
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23 references, click to expand
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Article Info
Journal
American journal of human genetics
Abbr.
Am J Hum Genet
ISSN
0002-9297
Published
1998-02-00
Pages
334-45
Language
English
Region
United States
NLM ID
0370475
PMCID
PMC1376883
Subset
IM
Grants
Wellcome Trust · United Kingdom
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