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Common and rare variant association analyses in amyotrophic lateral sclerosis identify 15 risk loci with distinct genetic architectures and neuron-specific biology.

van Rheenen W, van der Spek RAA, Bakker MK et al.

34873335 PubMed ID
GWAS Study Type
152268 Participants
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Chapter I

Publication Details

Comprehensive information about this research publication

Authors

VR
van Rheenen W
VD
van der Spek RAA
BM
Bakker MK
VV
van Vugt JJFA
HP
Hop PJ
ZR
Zwamborn RAJ
DK
de Klein N
WH
Westra HJ
BO
Bakker OB
DP
Deelen P
SG
Shireby G
HE
Hannon E
MM
Moisse M
BD
Baird D
RR
Restuadi R
DE
Dolzhenko E
DA
Dekker AM
GK
Gawor K
WH
Westeneng HJ
TG
Tazelaar GHP
VE
van Eijk KR
KM
Kooyman M
BR
Byrne RP
DM
Doherty M
HM
Heverin M
AK
Al Khleifat A
IA
Iacoangeli A
SA
Shatunov A
TN
Ticozzi N
CJ
Cooper-Knock J
SB
Smith BN
GM
Gromicho M
CS
Chandran S
PS
Pal S
MK
Morrison KE
SP
Shaw PJ
HJ
Hardy J
OR
Orrell RW
SM
Sendtner M
MT
Meyer T
BN
Başak N
VD
van der Kooi AJ
RA
Ratti A
FI
Fogh I
GC
Gellera C
LG
Lauria G
CS
Corti S
CC
Cereda C
SD
Sproviero D
DS
D'Alfonso S
SG
Sorarù G
SG
Siciliano G
FM
Filosto M
PA
Padovani A
CA
Chiò A
CA
Calvo A
MC
Moglia C
BM
Brunetti M
CA
Canosa A
GM
Grassano M
BE
Beghi E
PE
Pupillo E
LG
Logroscino G
NB
Nefussy B
OA
Osmanovic A
NA
Nordin A
LY
Lerner Y
ZM
Zabari M
GM
Gotkine M
BR
Baloh RH
BS
Bell S
VP
Vourc'h P
CP
Corcia P
CP
Couratier P
MS
Millecamps S
MV
Meininger V
SF
Salachas F
MP
Mora Pardina JS
AA
Assialioui A
RR
Rojas-García R
DP
Dion PA
RJ
Ross JP
LA
Ludolph AC
WJ
Weishaupt JH
BD
Brenner D
FA
Freischmidt A
BG
Bensimon G
BA
Brice A
DA
Durr A
PC
Payan CAM
SS
Saker-Delye S
WN
Wood NW
TS
Topp S
RR
Rademakers R
TL
Tittmann L
LW
Lieb W
FA
Franke A
RS
Ripke S
BA
Braun A
KJ
Kraft J
WD
Whiteman DC
OC
Olsen CM
UA
Uitterlinden AG
HA
Hofman A
RM
Rietschel M
CS
Cichon S
NM
Nöthen MM
AP
Amouyel P
TB
Traynor BJ
SA
Singleton AB
MN
Mitne Neto M
CR
Cauchi RJ
OR
Ophoff RA
WM
Wiedau-Pazos M
LC
Lomen-Hoerth C
VD
van Deerlin VM
GJ
Grosskreutz J
RA
Roediger A
GN
Gaur N
JA
Jörk A
BT
Barthel T
TE
Theele E
IB
Ilse B
SB
Stubendorff B
WO
Witte OW
SR
Steinbach R
HC
Hübner CA
GC
Graff C
BL
Brylev L
FV
Fominykh V
DV
Demeshonok V
AA
Ataulina A
RB
Rogelj B
KB
Koritnik B
ZJ
Zidar J
RM
Ravnik-Glavač M
GD
Glavač D
SZ
Stević Z
DV
Drory V
PM
Povedano M
BI
Blair IP
KM
Kiernan MC
BB
Benyamin B
HR
Henderson RD
FS
Furlong S
MS
Mathers S
MP
McCombe PA
NM
Needham M
NS
Ngo ST
NG
Nicholson GA
PR
Pamphlett R
RD
Rowe DB
SF
Steyn FJ
WK
Williams KL
MK
Mather KA
SP
Sachdev PS
HA
Henders AK
WL
Wallace L
DC
de Carvalho M
PS
Pinto S
PS
Petri S
WM
Weber M
RG
Rouleau GA
SV
Silani V
CC
Curtis CJ
BG
Breen G
GJ
Glass JD
BR
Brown RH
LJ
Landers JE
SC
Shaw CE
AP
Andersen PM
GE
Groen EJN
VE
van Es MA
PR
Pasterkamp RJ
FD
Fan D
GF
Garton FC
MA
McRae AF
DS
Davey Smith G
GT
Gaunt TR
EM
Eberle MA
MJ
Mill J
MR
McLaughlin RL
HO
Hardiman O
KK
Kenna KP
WN
Wray NR
TE
Tsai E
RH
Runz H
FL
Franke L
AA
Al-Chalabi A
VD
Van Damme P
VD
van den Berg LH
VJ
Veldink JH
Chapter II

Abstract

Summary of the research findings

Amyotrophic lateral sclerosis (ALS) is a fatal neurodegenerative disease with a lifetime risk of one in 350 people and an unmet need for disease-modifying therapies. We conducted a cross-ancestry genome-wide association study (GWAS) including 29,612 patients with ALS and 122,656 controls, which identified 15 risk loci. When combined with 8,953 individuals with whole-genome sequencing (6,538 patients, 2,415 controls) and a large cortex-derived expression quantitative trait locus (eQTL) dataset (MetaBrain), analyses revealed locus-specific genetic architectures in which we prioritized genes either through rare variants, short tandem repeats or regulatory effects. ALS-associated risk loci were shared with multiple traits within the neurodegenerative spectrum but with distinct enrichment patterns across brain regions and cell types. Of the environmental and lifestyle risk factors obtained from the literature, Mendelian randomization analyses indicated a causal role for high cholesterol levels. The combination of all ALS-associated signals reveals a role for perturbations in vesicle-mediated transport and autophagy and provides evidence for cell-autonomous disease initiation in glutamatergic neurons.

27,205 European ancestry cases, 110,881 European ancestry controls, 2,407 East Asian ancestry cases, 11,775 East Asian ancestry controls

Chapter III

Study Statistics

Key metrics and study information

152268
Total Participants
GWAS
Study Type
No
Replicated
European, East Asian
Ancestry
Russian Federation, Republic of Ireland, Portugal, Switzerland, Spain, Canada, Netherlands, Sweden, U.S., Belgium, Norway, Finland, Denmark, Italy, U.K., Slovenia, France, Serbia, Australia, Germany, China, Japan
Recruitment Country
Chapter IV

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