Modifying PCDH19 levels affects cortical interneuron migration.
PCDH19-CE
brain development
interneuron
medial ganglionic eminence
neurodevelopmental disorder
neuronal migration
Journal
Frontiers in neuroscience
ISSN: 1662-4548
Titre abrégé: Front Neurosci
Pays: Switzerland
ID NLM: 101478481
Informations de publication
Date de publication:
2022
2022
Historique:
received:
01
03
2022
accepted:
20
09
2022
entrez:
17
11
2022
pubmed:
18
11
2022
medline:
18
11
2022
Statut:
epublish
Résumé
PCDH19 is a transmembrane protein and member of the protocadherin family. It is encoded by the X-chromosome and more than 200 mutations have been linked to the neurodevelopmental PCDH-clustering epilepsy (PCDH19-CE) syndrome. A disturbed cell-cell contact that arises when random X-inactivation creates mosaic absence of PCDH19 has been proposed to cause the syndrome. Several studies have shown roles for PCDH19 in neuronal proliferation, migration, and synapse function, yet most of them have focused on cortical and hippocampal neurons. As epilepsy can also be caused by impaired interneuron migration, we studied the role of PCDH19 in cortical interneurons during embryogenesis. We show that cortical interneuron migration is affected by altering PCDH19 dosage by means of overexpression in brain slices and medial ganglionic eminence (MGE) explants. We also detect subtle defects when PCDH19 expression was reduced in MGE explants, suggesting that the dosage of PCDH19 is important for proper interneuron migration. We confirm this finding
Identifiants
pubmed: 36389226
doi: 10.3389/fnins.2022.887478
pmc: PMC9642031
doi:
Types de publication
Journal Article
Langues
eng
Pagination
887478Informations de copyright
Copyright © 2022 Pancho, Mitsogiannis, Aerts, Dalla Vecchia, Ebert, Geenen, Noterdaeme, Vanlaer, Stulens, Hulpiau, Staes, Van Roy, Dedecker, Schermer and Seuntjens.
Déclaration de conflit d'intérêts
The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.
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