Evidence map›Paper›PMID 28560159›Full record

ArticleNeuroImage. Clinical2017

Williams syndrome-specific neuroanatomical profile and its associations with behavioral features.

Chun Chieh Fan, Timothy T Brown, Hauke Bartsch, Joshua M Kuperman, Donald J Hagler, Andrew Schork, Yvonne Searcy, Ursula Bellugi, Eric Halgren, Anders M Dale

Open access · goldAbstract read
In one paragraph

Article in NeuroImage. Clinical, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers.

0numbers the graph read from it
0cells of the map it votes in
20citing papers in PubMed
2.1field-weighted citation impact, top 15% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

20 citing papers in PubMed, 30 citations in OpenAlex.

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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

10 authors at 2 institutions in 1 country.

Chun Chieh FanDepartment of Cognitive Science, University of California San Diego, 9500 Gilman Drive, La Jolla, CA 92093, USA; Center for Multimodal Imaging and Genetics, University of California San Diego, School of Medicine, 9452 Medical Center Drive, La Jolla, CA 92093, USA.
Timothy T BrownCenter for Multimodal Imaging and Genetics, University of California San Diego, School of Medicine, 9452 Medical Center Drive, La Jolla, CA 92093, USA; Department of Neurosciences, University of California San Diego, School of Medicine, 9500 Gilman Drive, La Jolla, CA 92037, USA; Center for Human Development, University of California San Diego, 9500 Gilman Dr., La Jolla, CA 92093, USA.
Hauke BartschCenter for Multimodal Imaging and Genetics, University of California San Diego, School of Medicine, 9452 Medical Center Drive, La Jolla, CA 92093, USA.
Joshua M KupermanCenter for Multimodal Imaging and Genetics, University of California San Diego, School of Medicine, 9452 Medical Center Drive, La Jolla, CA 92093, USA.
Donald J HaglerCenter for Multimodal Imaging and Genetics, University of California San Diego, School of Medicine, 9452 Medical Center Drive, La Jolla, CA 92093, USA; Department of Radiology, University of California San Diego, School of Medicine, 9500 Gilman Drive, La Jolla, CA 92037, USA.
Andrew SchorkDepartment of Cognitive Science, University of California San Diego, 9500 Gilman Drive, La Jolla, CA 92093, USA; Center for Multimodal Imaging and Genetics, University of California San Diego, School of Medicine, 9452 Medical Center Drive, La Jolla, CA 92093, USA.
Yvonne SearcyLaboratory for Cognitive Neuroscience, Salk Institute, La Jolla, CA 92037, USA.
Ursula BellugiLaboratory for Cognitive Neuroscience, Salk Institute, La Jolla, CA 92037, USA.
Eric HalgrenDepartment of Neurosciences, University of California San Diego, School of Medicine, 9500 Gilman Drive, La Jolla, CA 92037, USA; Department of Radiology, University of California San Diego, School of Medicine, 9500 Gilman Drive, La Jolla, CA 92037, USA; Center for Human Brain Activity Mapping, University of California San Diego, School of Medicine, 3510 Dunhill Street, San Diego, CA 92121, USA. Electronic address: ehalgren@ucsd.edu.
Anders M DaleDepartment of Cognitive Science, University of California San Diego, 9500 Gilman Drive, La Jolla, CA 92093, USA; Center for Multimodal Imaging and Genetics, University of California San Diego, School of Medicine, 9452 Medical Center Drive, La Jolla, CA 92093, USA; Department of Neurosciences, University of California San Diego, School of Medicine, 9500 Gilman Drive, La Jolla, CA 92037, USA; Department of Radiology, University of California San Diego, School of Medicine, 9500 Gilman Drive, La Jolla, CA 92037, USA. Electronic address: amdale@ucsd.edu.
University of California San Diego · USSalk Institute for Biological Studies · US

Funding

VARIANTS OF FOCAL BRAIN INJURYP50NS022343 · NINDS · UNIVERSITY OF CALIFORNIA, SAN DIEGO · PI TRAUNER, DORIS A · 1985 to 2012
$15.1M
Examination of Neurobehavioral Development Using the PING Data ResourceR01DA038958 · NIDA · UNIVERSITY OF CALIFORNIA, SAN DIEGO · PI AKSHOOMOFF, NATACHA, BROWN, TIMOTHY TODD · 2014 to 2016
$830k
NIDA NIH HHS R01 DA038958NINDS NIH HHS P50 NS022343
6 · The paper itself

Abstract

Williams Syndrome (WS) is a rare genetic disorder with unique behavioral features. Yet the rareness of WS has limited the number and type of studies that can be conducted in which inferences are made about how neuroanatomical abnormalities mediate behaviors. In this study, we extracted a WS-specific neuroanatomical profile from structural magnetic resonance imaging (MRI) measurements and tested its association with behavioral features of WS. Using a WS adult cohort (22 WS, 16 healthy controls), we modeled a sparse representation of a WS-specific neuroanatomical profile. The predictive performances are robust within the training cohort (10-fold cross-validation, AUC = 1.0) and accurately identify all WS individuals in an independent child WS cohort (seven WS, 59 children with diverse developmental status, AUC = 1.0). The WS-specific neuroanatomical profile includes measurements in the orbitofrontal cortex, superior parietal cortex, Sylvian fissures, and basal ganglia, and variability within these areas related to the underlying size of hemizygous deletion in patients with partial deletions. The profile intensity mediated the overall cognitive impairment as well as personality features related to hypersociability. Our results imply that the unique behaviors in WS were mediated through the constellation of abnormalities in cortical-subcortical circuitry consistent in child WS and adult WS. The robustness of the derived WS-specific neuroanatomical profile also demonstrates the potential utility of our approach in both clinical and research applications.

Indexed as

AdolescentAdultBasal GangliaCerebral CortexCognitive DysfunctionCohort StudiesDevelopmental DisabilitiesFemaleHumansMagnetic Resonance ImagingMaleSocial PerceptionWilliams SyndromeYoung AdultMagnetic resonance imagingNeuroanatomySocial cognitionWilliams Syndrome

Identifiers

PMID28560159
PMCPMC5443907
OpenAlexW2616276324

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
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Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.