Genome-wide association of familial late-onset Alzheimer's disease replicates BIN1 and CLU and nominates CUGBP2 in interaction with APOE.

Late-onset Alzheimer's disease (LOAD) is the most common form of dementia in the elderly. The National Institute of Aging-Late Onset Alzheimer's Disease Family Study and the National Cell Repository for Alzheimer's Disease conducted a joint genome-wide association study (GWAS) of mult...

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Main Authors: Ellen M Wijsman, Nathan D Pankratz, Yoonha Choi, Joseph H Rothstein, Kelley M Faber, Rong Cheng, Joseph H Lee, Thomas D Bird, David A Bennett, Ramon Diaz-Arrastia, Alison M Goate, Martin Farlow, Bernardino Ghetti, Robert A Sweet, Tatiana M Foroud, Richard Mayeux, NIA-LOAD/NCRAD Family Study Group
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Language:English
Published: Public Library of Science (PLoS) 2011-02-01
Series:PLoS Genetics
Online Access:https://journals.plos.org/plosgenetics/article/file?id=10.1371/journal.pgen.1001308&type=printable
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author Ellen M Wijsman
Nathan D Pankratz
Yoonha Choi
Joseph H Rothstein
Kelley M Faber
Rong Cheng
Joseph H Lee
Thomas D Bird
David A Bennett
Ramon Diaz-Arrastia
Alison M Goate
Martin Farlow
Bernardino Ghetti
Robert A Sweet
Tatiana M Foroud
Richard Mayeux
NIA-LOAD/NCRAD Family Study Group
author_facet Ellen M Wijsman
Nathan D Pankratz
Yoonha Choi
Joseph H Rothstein
Kelley M Faber
Rong Cheng
Joseph H Lee
Thomas D Bird
David A Bennett
Ramon Diaz-Arrastia
Alison M Goate
Martin Farlow
Bernardino Ghetti
Robert A Sweet
Tatiana M Foroud
Richard Mayeux
NIA-LOAD/NCRAD Family Study Group
author_sort Ellen M Wijsman
collection DOAJ
description Late-onset Alzheimer's disease (LOAD) is the most common form of dementia in the elderly. The National Institute of Aging-Late Onset Alzheimer's Disease Family Study and the National Cell Repository for Alzheimer's Disease conducted a joint genome-wide association study (GWAS) of multiplex LOAD families (3,839 affected and unaffected individuals from 992 families plus additional unrelated neurologically evaluated normal subjects) using the 610 IlluminaQuad panel. This cohort represents the largest family-based GWAS of LOAD to date, with analyses limited here to the European-American subjects. SNPs near APOE gave highly significant results (e.g., rs2075650, p = 3.2×10(-81)), but no other genome-wide significant evidence for association was obtained in the full sample. Analyses that stratified on APOE genotypes identified SNPs on chromosome 10p14 in CUGBP2 with genome-wide significant evidence for association within APOE ε4 homozygotes (e.g., rs201119, p = 1.5×10(-8)). Association in this gene was replicated in an independent sample consisting of three cohorts. There was evidence of association for recently-reported LOAD risk loci, including BIN1 (rs7561528, p = 0.009 with, and p = 0.03 without, APOE adjustment) and CLU (rs11136000, p = 0.023 with, and p = 0.008 without, APOE adjustment), with weaker support for CR1. However, our results provide strong evidence that association with PICALM (rs3851179, p = 0.69 with, and p = 0.039 without, APOE adjustment) and EXOC3L2 is affected by correlation with APOE, and thus may represent spurious association. Our results indicate that genetic structure coupled with ascertainment bias resulting from the strong APOE association affect genome-wide results and interpretation of some recently reported associations. We show that a locus such as APOE, with large effects and strong association with disease, can lead to samples that require appropriate adjustment for this locus to avoid both false positive and false negative evidence of association. We suggest that similar adjustments may also be needed for many other large multi-site studies.
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publisher Public Library of Science (PLoS)
record_format Article
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spelling doaj-art-bcc36e189ec444929a0949cdbbbdb4292025-08-20T02:33:55ZengPublic Library of Science (PLoS)PLoS Genetics1553-73901553-74042011-02-0172e100130810.1371/journal.pgen.1001308Genome-wide association of familial late-onset Alzheimer's disease replicates BIN1 and CLU and nominates CUGBP2 in interaction with APOE.Ellen M WijsmanNathan D PankratzYoonha ChoiJoseph H RothsteinKelley M FaberRong ChengJoseph H LeeThomas D BirdDavid A BennettRamon Diaz-ArrastiaAlison M GoateMartin FarlowBernardino GhettiRobert A SweetTatiana M ForoudRichard MayeuxNIA-LOAD/NCRAD Family Study GroupLate-onset Alzheimer's disease (LOAD) is the most common form of dementia in the elderly. The National Institute of Aging-Late Onset Alzheimer's Disease Family Study and the National Cell Repository for Alzheimer's Disease conducted a joint genome-wide association study (GWAS) of multiplex LOAD families (3,839 affected and unaffected individuals from 992 families plus additional unrelated neurologically evaluated normal subjects) using the 610 IlluminaQuad panel. This cohort represents the largest family-based GWAS of LOAD to date, with analyses limited here to the European-American subjects. SNPs near APOE gave highly significant results (e.g., rs2075650, p = 3.2×10(-81)), but no other genome-wide significant evidence for association was obtained in the full sample. Analyses that stratified on APOE genotypes identified SNPs on chromosome 10p14 in CUGBP2 with genome-wide significant evidence for association within APOE ε4 homozygotes (e.g., rs201119, p = 1.5×10(-8)). Association in this gene was replicated in an independent sample consisting of three cohorts. There was evidence of association for recently-reported LOAD risk loci, including BIN1 (rs7561528, p = 0.009 with, and p = 0.03 without, APOE adjustment) and CLU (rs11136000, p = 0.023 with, and p = 0.008 without, APOE adjustment), with weaker support for CR1. However, our results provide strong evidence that association with PICALM (rs3851179, p = 0.69 with, and p = 0.039 without, APOE adjustment) and EXOC3L2 is affected by correlation with APOE, and thus may represent spurious association. Our results indicate that genetic structure coupled with ascertainment bias resulting from the strong APOE association affect genome-wide results and interpretation of some recently reported associations. We show that a locus such as APOE, with large effects and strong association with disease, can lead to samples that require appropriate adjustment for this locus to avoid both false positive and false negative evidence of association. We suggest that similar adjustments may also be needed for many other large multi-site studies.https://journals.plos.org/plosgenetics/article/file?id=10.1371/journal.pgen.1001308&type=printable
spellingShingle Ellen M Wijsman
Nathan D Pankratz
Yoonha Choi
Joseph H Rothstein
Kelley M Faber
Rong Cheng
Joseph H Lee
Thomas D Bird
David A Bennett
Ramon Diaz-Arrastia
Alison M Goate
Martin Farlow
Bernardino Ghetti
Robert A Sweet
Tatiana M Foroud
Richard Mayeux
NIA-LOAD/NCRAD Family Study Group
Genome-wide association of familial late-onset Alzheimer's disease replicates BIN1 and CLU and nominates CUGBP2 in interaction with APOE.
PLoS Genetics
title Genome-wide association of familial late-onset Alzheimer's disease replicates BIN1 and CLU and nominates CUGBP2 in interaction with APOE.
title_full Genome-wide association of familial late-onset Alzheimer's disease replicates BIN1 and CLU and nominates CUGBP2 in interaction with APOE.
title_fullStr Genome-wide association of familial late-onset Alzheimer's disease replicates BIN1 and CLU and nominates CUGBP2 in interaction with APOE.
title_full_unstemmed Genome-wide association of familial late-onset Alzheimer's disease replicates BIN1 and CLU and nominates CUGBP2 in interaction with APOE.
title_short Genome-wide association of familial late-onset Alzheimer's disease replicates BIN1 and CLU and nominates CUGBP2 in interaction with APOE.
title_sort genome wide association of familial late onset alzheimer s disease replicates bin1 and clu and nominates cugbp2 in interaction with apoe
url https://journals.plos.org/plosgenetics/article/file?id=10.1371/journal.pgen.1001308&type=printable
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