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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