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Common variants at SCN5A-SCN10A and HEY2 are associated with Brugada syndrome, a rare disease with high risk of sudden cardiac death.

Bezzina CR, Barc J, Mizusawa Y et al.

23872634 PubMed ID
GWAS Study Type
4051 Participants
1,368 Views
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Chapter I

Publication Details

Comprehensive information about this research publication

Authors

BC
Bezzina CR
BJ
Barc J
MY
Mizusawa Y
RC
Remme CA
GJ
Gourraud JB
SF
Simonet F
VA
Verkerk AO
SP
Schwartz PJ
CL
Crotti L
DF
Dagradi F
GP
Guicheney P
FV
Fressart V
LA
Leenhardt A
AC
Antzelevitch C
BS
Bartkowiak S
BM
Borggrefe M
SR
Schimpf R
SE
Schulze-Bahr E
ZS
Zumhagen S
BE
Behr ER
BR
Bastiaenen R
TJ
Tfelt-Hansen J
OM
Olesen MS
KS
Kääb S
BB
Beckmann BM
WP
Weeke P
WH
Watanabe H
EN
Endo N
MT
Minamino T
HM
Horie M
OS
Ohno S
HK
Hasegawa K
MN
Makita N
NA
Nogami A
SW
Shimizu W
AT
Aiba T
FP
Froguel P
BB
Balkau B
LO
Lantieri O
TM
Torchio M
WC
Wiese C
WD
Weber D
WR
Wolswinkel R
CR
Coronel R
BB
Boukens BJ
BS
Bézieau S
CE
Charpentier E
CS
Chatel S
DA
Despres A
GF
Gros F
KF
Kyndt F
LS
Lecointe S
LP
Lindenbaum P
PV
Portero V
VJ
Violleau J
GM
Gessler M
TH
Tan HL
RD
Roden DM
CV
Christoffels VM
LM
Le Marec H
WA
Wilde AA
PV
Probst V
SJ
Schott JJ
DC
Dina C
RR
Redon R
Chapter II

Abstract

Summary of the research findings

Brugada syndrome is a rare cardiac arrhythmia disorder, causally related to SCN5A mutations in around 20% of cases. Through a genome-wide association study of 312 individuals with Brugada syndrome and 1,115 controls, we detected 2 significant association signals at the SCN10A locus (rs10428132) and near the HEY2 gene (rs9388451). Independent replication confirmed both signals (meta-analyses: rs10428132, P = 1.0 × 10(-68); rs9388451, P = 5.1 × 10(-17)) and identified one additional signal in SCN5A (at 3p21; rs11708996, P = 1.0 × 10(-14)). The cumulative effect of the three loci on disease susceptibility was unexpectedly large (Ptrend = 6.1 × 10(-81)). The association signals at SCN5A-SCN10A demonstrate that genetic polymorphisms modulating cardiac conduction can also influence susceptibility to cardiac arrhythmia. The implication of association with HEY2, supported by new evidence that Hey2 regulates cardiac electrical activity, shows that Brugada syndrome may originate from altered transcriptional programming during cardiac development. Altogether, our findings indicate that common genetic variation can have a strong impact on the predisposition to rare diseases.

312 European ancestry cases, 1,115 European ancestry controls

Chapter III

Study Statistics

Key metrics and study information

4051
Total Participants
GWAS
Study Type
Yes
Replicated
594 European ancestry cases, 806 European ancestry controls, 208 Japanese ancestry cases, 1,1016 Japanese controls
Replication Participants
European, East Asian
Ancestry
Italy, Netherlands, Germany, U.K., France, Denmark, Japan
Recruitment Country
Chapter IV

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