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Altered structural connectivity networks in a mouse model of complete and partial dysgenesis of the corpus callosum

Edwards, Timothy J. and Fenlon, Laura R. and Dean, Ryan J. and Bunt, Jens and Sherr, Elliott H. and Richards, Linda J. (2020) Altered structural connectivity networks in a mouse model of complete and partial dysgenesis of the corpus callosum. NeuroImage, 217 . Art. No. 116868. ISSN 1053-8119. doi:10.1016/j.neuroimage.2020.116868.

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Corpus callosum dysgenesis (CCD) describes a collection of brain malformations in which the main fiber tract connecting the two hemispheres is either absent (complete CCD, or ‘agenesis of the corpus callosum’) or reduced in size (partial CCD). Humans with these neurodevelopmental disorders have a wide range of cognitive outcomes, including seemingly preserved features of interhemispheric communication in some cases. However, the structural substrates that could underlie this variability in outcome remain to be fully elucidated. Here, for the first time, we characterize the global brain connectivity of a mouse model of complete and partial CCD. We demonstrate features of structural brain connectivity that model those predicted in humans with CCD, including Probst bundles in complete CCD and heterotopic sigmoidal connections in partial CCD. Crucially, we also histologically validate the recently predicted ectopic sigmoid bundle present in humans with partial CCD, validating the utility of this mouse model for fine anatomical studies of this disorder. Taken together, this work describes a mouse model of altered structural connectivity in variable severity CCD and forms a foundation for future studies investigating the function and mechanisms of development of plastic tracts in developmental disorders of brain connectivity.

Item Type:Article
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URLURL TypeDescription
https://www.irc5.orgRelated ItemInternational Research Consortium for the Corpus Callosum and Cerebral Connectivity
Fenlon, Laura R.0000-0002-5132-2419
Other Contributors:
ContributionOther Contributors NameIdentifierPersonID (may be blank)
CollaboratorPaul, Lynn K.Paul-Lynn-KUNSPECIFIED
Additional Information:© 2020 The Author(s). Published by Elsevier Inc. This is an open access article under the CC BY license ( Received 31 October 2019, Revised 13 April 2020, Accepted 20 April 2020, Available online 29 April 2020. We thank The University of Queensland Biological Resources and Queensland Brain Institute animal team for animal support and the Queensland Brain Institute Microscopy team for imaging support. We thank Dr. Nyoman Kurniawan for his assistance in acquiring MRI scans at the Centre for Advanced Imaging at The University of Queensland, a National Collaborative Research Infrastructure Strategy funded facility. We thank Prof. Alan Connelly, Dr. Yonghui Li and Dr. Cirong Liu for initial contributions to this project. We thank Prof. Fernanda Tovar-Moll and Dr. Diego Szczupak for input on the manuscript. Finally, we thank the International Research Consortium for the Corpus Callosum and Cerebral Connectivity (IRC5, researchers for discussions and input. The IRC5 researchers are Glena Andrews, Emanuela Argilli, Filippo Arrigoni, Nadia Bahi-Buisson, Rodrigo Basilio, Anais Bellon, Tania Attie-Bitach, Nathalie Boddaert, Rhonda Booth, Lucille Boutard, Ivanei Bramati, Warren Brown, Jens Bunt, Alain Chedotal, Ryan Dean, Christel Depienne, Jessica Dubois, Timothy Edwards, Laura Fenlon, Catherine Garel, Sonia Garel, Lucas Gemal, Kim Giraudat, Sarah Glatter, Orit Glenn, Juliette Godin, Solveiq Heide, Delphine Heron, Rujii Jiang, Bethany Johnson-Kern, David Kahn, Gregor Kasprian, Dorit Kliemann, Emmanuelle Lacaze, Roberto Lent, Richard Leventer, Marco Leyton, Paul Lockhart, Zoran Lynton, Paul Lockhart, George McGillivray, Theo Marins, Ashley Marsh, Bruno Melo, Cyril Mignot, Anne Elodie Millisher, Ching Moey, Jorge Moll, Myriam Monteiro, Marie-Laur Moutard, Pratik Mukherjee, Remya Nair, Marta Nieto-Lopez, Lynn K Paul, Alessandra Pierrani, Ferechte Encha-Razavi, Linda J Richards, Gail Robinson, Romina Romaniello, Rinuba Rinabuekki,Tally Lerman-Sagie, Laurent Salomon, Elliott H Sherr, Vanessa Siffredi, Myrtille Spentchian, Diego Szcupak, Victor Tarabykin, Sophie Thomas, Fernanda Tovar-Moll, Jasmin Turner, Stephanie Valence, Mark Walterfang, Natalia Rich-Wimmer, Alice Wright, Binnaz Yalcin. This work was supported by National Health and Medical Research Council (NHMRC) grants 1126053 and 1124411 and NIH grant NS058721. TJE was supported by a University of Queensland Research Scholarship. LRF was supported by a UQ Development Fellowship and NHMRC Investigator Grant GNT1175825. RJD was supported by Brain Injured Childrens After-Care Recovery Endeavours (BICARE) Inc. LJR was supported by NHMRC Principal Research Fellowship GNT1120615. CRediT authorship contribution statement: Timothy J. Edwards: Conceptualization, Data curation, Formal analysis, Investigation, Methodology, Writing - original draft. Laura R. Fenlon: Data curation, Formal analysis, Funding acquisition, Investigation, Methodology, Writing - original draft. Ryan J. Dean: Data curation, Formal analysis, Methodology, Writing - original draft. Jens Bunt: Conceptualization, Supervision, Writing - review & editing. Elliott H. Sherr: Conceptualization, Funding acquisition, Supervision, Writing - review & editing. Linda J. Richards: Conceptualization, Formal analysis, Project administration, Resources, Supervision, Writing - review & editing, Funding acquisition. The authors declare no competing financial interests.
Funding AgencyGrant Number
National Health and Medical Research Council (NHMRC)1126053
National Health and Medical Research Council (NHMRC)1124411
University of QueenslandUNSPECIFIED
National Health and Medical Research Council (NHMRC)GNT1175825
Brain Injured Childrens After-Care Recovery Endeavours (BICARE) Inc.UNSPECIFIED
National Health and Medical Research Council (NHMRC)GNT1120615
Subject Keywords:Corpus callosum; Corpus callosum dysgenesis; Structural connectome; Cortical development; Brain plasticity; Neurodevelopmental disorders
Record Number:CaltechAUTHORS:20210302-101629757
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Official Citation:Timothy J. Edwards, Laura R. Fenlon, Ryan J. Dean, Jens Bunt, Elliott H. Sherr, Linda J. Richards, Altered structural connectivity networks in a mouse model of complete and partial dysgenesis of the corpus callosum, NeuroImage, Volume 217, 2020, 116868, ISSN 1053-8119, (
Usage Policy:No commercial reproduction, distribution, display or performance rights in this work are provided.
ID Code:108268
Deposited By: Tony Diaz
Deposited On:02 Mar 2021 18:28
Last Modified:16 Nov 2021 19:10

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