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PMID: 9973294 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, Non-P.H.S. Research Support, U.S. Gov't, P.H.S.

Novel locus for autosomal dominant hereditary spastic paraplegia, on chromosome 8q.

American journal of human genetics ·Vol. 64 ·No. 2 ·1999-02-00 ·Pages 563-9

Hedera P, Rainier S, Alvarado D, Zhao X, Williamson J, Otterud B, Leppert M, Fink JK

Abstract

Hereditary spastic paraplegia (HSP) is a clinically and genetically heterogeneous group of disorders characterized by insidiously progressive spastic weakness in the legs. Genetic loci for autosomal dominant HSP exist on chromosomes 2p, 14q, and 15q. These loci are excluded in 45% of autosomal dominant HSP kindreds, indicating the presence of additional loci for autosomal dominant HSP. We analyzed a Caucasian kindred with autosomal dominant HSP and identified tight linkage between the disorder and microsatellite markers on chromosome 8q (maximum two-point LOD score 5.51 at recombination fraction 0). Our results clearly establish the existence of a locus for autosomal dominant HSP on chromosome 8q23-24. Currently this locus spans 6.2 cM between D8S1804 and D8S1774 and includes several potential candidate genes. Identifying this novel HSP locus on chromosome 8q23-24 will facilitate discovery of this HSP gene, improve genetic counseling for families with linkage to this locus, and extend our ability to correlate clinical features with different HSP loci.

MeSH Terms
Adult Chromosomes, Human, Pair 8 Female Genes, Dominant Genetic Linkage Genetic Testing Humans Male Middle Aged Pedigree Spastic Paraplegia, Hereditary/genetics,physiopathology
Authors & Affiliations
8 authors, click to expand affiliations / ORCID
Hedera P
Department of Neurology, University of Michigan, Ann Arbor, MI 48109-0940, USA.
Rainier S
Alvarado D
Zhao X
Williamson J
Otterud B
Leppert M
Fink J K
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Article Info
Journal
American journal of human genetics
Abbr.
Am J Hum Genet
ISSN
0002-9297
Published
1999-02-00
Pages
563-9
Language
English
Region
United States
NLM ID
0370475
PMCID
PMC1377766
Subset
IM
Grants
NINDS NIH HHS · R01NS33645 · United States
NINDS NIH HHS · R01NS36177 · United States
NINDS NIH HHS · T32NS07222 · United States
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