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@ARTICLE{Sonderby:848364,
author = {Sonderby, I. E. and Gustafsson, O. and Doan, N. T. and
Hibar, D. P. and Martin-Brevet, S. and Westley, L. T. and
Jacquemont, S. and Djurovic, S. and Stefansson, H. and
Stefansson, K. and Thompson, P. M. and Andreassen, O. A. and
Cichon, Sven},
title = {{D}ose response of the 16p11.2 digital copy number on
intracranial volume and basal ganglia},
journal = {Molecular psychiatry},
volume = {25},
issn = {1359-4184},
address = {London},
publisher = {Macmillan},
reportid = {FZJ-2018-03605},
pages = {584–602},
year = {2020},
abstract = {Carriers of large recurrent copy number variants (CNVs)
have a higher risk of developing neurodevelopmental
disorders. The 16p11.2 distal CNV predisposes carriers to
e.g., autism spectrum disorder and schizophrenia. We
compared subcortical brain volumes of 12 16p11.2 distal
deletion and 12 duplication carriers to 6882 non-carriers
from the large-scale brain Magnetic Resonance Imaging
collaboration, ENIGMA-CNV. After stringent CNV calling
procedures, and standardized FreeSurfer image analysis, we
found negative dose-response associations with copy number
on intracranial volume and on regional caudate, pallidum and
putamen volumes (β = −0.71 to −1.37;
P < 0.0005). In an independent sample, consistent
results were obtained, with significant effects in the
pallidum (β = −0.95, P = 0.0042). The two data
sets combined showed significant negative dose-response for
the accumbens, caudate, pallidum, putamen and ICV
(P = 0.0032, 8.9 × 10−6, 1.7 × 10−9,
3.5 × 10−12 and 1.0 × 10−4, respectively).
Full scale IQ was lower in both deletion and duplication
carriers compared to non-carriers. This is the first brain
MRI study of the impact of the 16p11.2 distal CNV, and we
demonstrate a specific effect on subcortical brain
structures, suggesting a neuropathological pattern
underlying the neurodevelopmental syndromes.},
cin = {INM-1},
ddc = {610},
cid = {I:(DE-Juel1)INM-1-20090406},
pnm = {571 - Connectivity and Activity (POF3-571)},
pid = {G:(DE-HGF)POF3-571},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:30283035},
UT = {WOS:000516569700011},
doi = {10.1038/s41380-018-0118-1},
url = {https://juser.fz-juelich.de/record/848364},
}