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@ARTICLE{Sonderby:894519,
author = {Sonderby, IE and Van der Meer, D. and Moreau, C. and
Kaufmann, T. and Bragi Walters, G. and Ellegaard, M. and
Abdellaoui, A. and Ames, D. and Amunts, K. and Andersson, M.
and Armstrong, NJ and Bernard, M. and Blackburn, NB and
Blangero, J. and Boomsma, DI and Brodaty, H. and Brouwer, RM
and Bülow, R. and Boen, R. and Cahn, W. and Calhoun, VD and
Caspers, S. and Ching, CRK and Cichon, S. and Cuifolini, S.
and Crespo-Facorro, B. and Curran, JE and Dale, AM and
Dalvie, S. and Dazzan, P. and De Geus, EJC and De Zubicaray,
GI and De Zwarte, SMC and Desrivieres, S. and Doherty, JL
and Donohoe, G. and Draganski, B. and Ehrlich, S. and
Eising, E. and Espeseth, T. and Fejgin, K. and Fisher, SE
and Fladby, T. and Frei, O. and Frouin, V. and Fukunaga, M.
and Gareau, T. and Ge, T. and Glahn, DC and Grabe, HJ and
Groenewold, NA and Gustafsson, O. and Haavik, J. and Haberg,
AK and Hall, J. and Hashimoto, R. and Hehir-Kwa, JY and
Hibar, DP and Hillegers, MHJ and Hoffmann, P. and Holleran,
L. and Holmes, AJ and Homuth, G. and Hottenga, JJ and
Hulshoff, Pol, HE and Ikeda, M. and Jahanshad, N. and
Jockwitz, C. and Johansson, S. and Jönsson, EG and
Jorgensen, NR and Kikuchi, M. and Knowles, EEM and Kumar, K.
and Le Hellard, S. and Leu, C. and Linden, DE and Liu, J.
and Lundervold, A. and Lundervold, AJ and Maillard, AM and
Martin, NG and Martin-Brevet, S. and Mather, KA and Mathias,
SR and McMahon, KL and McRae, AF and Medland, SE and
Meyer-Lindenberg, A. and Moberget, T. and Modenato, C. and
Monereo, Sánchez, J and Morris, DW and Mühleisen, TW and
Murray, RM and Nielsen, J. and Nordvik, JE and Nyberg, L.
and Olde Loohuis, LM and Ophoff, RA and Owen, MJ and Paus,
T. and Pausova, Z. and Peralta, JM and Pike, B. and Prieto,
C. and Quinlan, EB and Reinbold, CS and Reis, Marques, T and
Rucker, JJH and Sachdev, PS and Sando, SB and Schofield, PR
and Schork, AJ and Schumann, G. and Shin, J. and Shumskaya,
E. and Silva, AI and Sisodiya, SM and Steen, VM and Stein,
DJ and Strike, LT and Suzuki, IK and Tamnes, CK and Teumer,
A. and Thalamuthu, A. and Tordesillas-Gutiérrez, D. and
Uhlmann, A. and Úlfarsson, MÖ and van´t Ent, D. and von
den Bree, MBM and Vanderhaeghen, P. and Vassos, E. and Wen,
W. and Wittfeld, K. and Wright, MJ and Agartz, I. and
Djurovic, S. and Westlye, LT and Stefánsson, H. and
Stefánsson, K. and Jacquemont, S. and Thompson, PM and
Andreassen, OA},
title = {1q21.1 distal copy number variants are associated with
cerebral and cognitive alterations in humans},
journal = {Translational Psychiatry},
volume = {11},
number = {1},
issn = {2158-3188},
address = {London},
publisher = {Nature Publishing Group},
reportid = {FZJ-2021-03255},
pages = {182},
year = {2021},
abstract = {Low-frequency 1q21.1 distal deletion and duplication copy
number variant (CNV) carriers are predisposed to multiple
neurodevelopmental disorders, including schizophrenia,
autism and intellectual disability. Human carriers display a
high prevalence of micro- and macrocephaly in deletion and
duplication carriers, respectively. The underlying brain
structural diversity remains largely unknown. We
systematically called CNVs in 38 cohorts from the
large-scale ENIGMA-CNV collaboration and the UK Biobank and
identified 28 1q21.1 distal deletion and 22 duplication
carriers and 37,088 non-carriers $(48\%$ male) derived from
15 distinct magnetic resonance imaging scanner sites. With
standardized methods, we compared subcortical and cortical
brain measures (all) and cognitive performance (UK Biobank
only) between carrier groups also testing for mediation of
brain structure on cognition. We identified positive dosage
effects of copy number on intracranial volume (ICV) and
total cortical surface area, with the largest effects in
frontal and cingulate cortices, and negative dosage effects
on caudate and hippocampal volumes. The carriers displayed
distinct cognitive deficit profiles in cognitive tasks from
the UK Biobank with intermediate decreases in duplication
carriers and somewhat larger in deletion carriers—the
latter potentially mediated by ICV or cortical surface area.
These results shed light on pathobiological mechanisms of
neurodevelopmental disorders, by demonstrating gene dose
effect on specific brain structures and effect on cognitive
function.},
cin = {INM-1},
ddc = {610},
cid = {I:(DE-Juel1)INM-1-20090406},
pnm = {5251 - Multilevel Brain Organization and Variability
(POF4-525) / 571 - Connectivity and Activity (POF3-571) /
HBP SGA3 - Human Brain Project Specific Grant Agreement 3
(945539)},
pid = {G:(DE-HGF)POF4-5251 / G:(DE-HGF)POF3-571 /
G:(EU-Grant)945539},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:33753722},
UT = {WOS:000632961500003},
doi = {10.1038/s41398-021-01213-0},
url = {https://juser.fz-juelich.de/record/894519},
}