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PMID: 9629852 Published · ppublish English Journal Article Research Support, U.S. Gov't, P.H.S.

Tau is a candidate gene for chromosome 17 frontotemporal dementia.

Annals of neurology ·Vol. 43 ·No. 6 ·1998-06-00 ·Pages 815-25

Poorkaj P, Bird TD, Wijsman E, Nemens E, Garruto RM, Anderson L, Andreadis A, Wiederholt WC, Raskind M, Schellenberg GD

Abstract

Frontotemporal dementia with parkinsonism, chromosome 17 type (FTDP-17), a recently defined disease entity, is clinically characterized by personality changes sometimes associated with psychosis, hyperorality, and diminished speech output, disturbed executive function and nonfluent aphasia, bradykinesia, and rigidity. Neuropathological changes include frontotemporal atrophy often associated with atrophy of the basal ganglia, substantia nigra, and amygdala. Neurofibrillary tangles (NFTs) are seen in some but not all families. Inheritance is autosomal dominant and the gene has been regionally localized to 17q21-22 in a 2- to 4-centimorgan (cM) region flanked by markers D17S800 and D17S791. The gene for tau, the primary component of NFTs, is located in the same region of chromosome 17. Tau was evaluated as a candidate gene. Physical mapping studies place tau within 2 megabases or less of D17S791, but it is probably outside the D17S800-D17S791 FTDP-17 interval. DNA sequence analysis of tau coding regions in affected subjects from two FTDP-17 families revealed nine DNA sequence variants, eight of which were also identified in controls and are thus polymorphisms. A ninth variant (Val279Met) was found in one FTDP-17 family but not in the second FTDP-17 family. Three lines of evidence indicate that the Val279Met change is an FTDP-17 causative mutation. First, the mutation site is highly conserved, and a normal valine is found at this position in all three tau interrepeat sequences and in other microtubule associated protein tau homologues. Second, the mutation co-segregates with the disease in family A. Third, the mutation is not found in normal controls.

MeSH Terms
Chromosome Mapping Chromosomes, Human, Pair 17 DNA Primers Dementia/genetics Family Health Female Frontal Lobe/physiopathology Genetic Linkage Humans Male Molecular Sequence Data Pedigree Polymorphism, Genetic Sequence Homology, Amino Acid Temporal Lobe/physiopathology tau Proteins/genetics
Chemicals
DNA Primers tau Proteins
Authors & Affiliations
10 authors, click to expand affiliations / ORCID
Poorkaj P
Geriatric Research Education Clinical Center, Veterans Affairs Puget Sound Health Care System, Division of Gerontology, University of Washington, Seattle 98108, USA.
Bird T D
Wijsman E
Nemens E
Garruto R M
Anderson L
Andreadis A
Wiederholt W C
Raskind M
Schellenberg G D
Article Info
Journal
Annals of neurology
Abbr.
Ann Neurol
ISSN
0364-5134
Published
1998-06-00
Pages
815-25
Language
English
Region
United States
NLM ID
7707449
Subset
IM
Grants
NIA NIH HHS · AG05136 · United States
NIA NIH HHS · AG1176-03 · United States
NIA NIH HHS · P01 AG14382 · United States
Corrections
ErratumIn
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