MCF2 is linked to a complex perisylvian syndrome and affects cortical lamination
Aude Molinard‐ChenuiD, Joël FlussiD, Sacha LaurentiD, Méryle Laurent, Michel GuipponiiD, Alexandre G. DayeriD
Abstract
The combination of congenital bilateral perisylvian syndrome (CBPS) with lower motor neuron dysfunction is unusual and suggests a potential common genetic insult affecting basic neurodevelopmental processes. Here we identify a putatively pathogenic missense mutation in the MCF2 gene in a boy with CBPS. Using in utero electroporation to genetically manipulate cortical neurons during corticogenesis, we demonstrate that the mouse Mcf2 gene controls the embryonic migration of cortical projection neurons. Strikingly, we find that the CBPS-associated MCF2 mutation impairs cortical laminar positioning, supporting the hypothesis that alterations in the process of embryonic neuronal migration can lead to rare cases of CBPS.
02 /Specification & Classification
Click any tag to browse matching papers- Citations
- 6
- FWCI
- 0.26
- Field percentile
- top 38%
- Organism
- Approach
Reproducibility ledger
Nobody has reported trying to reproduce this paper yet. Did you replicate an experiment or run the code? Your report saves the next lab a week.
Sign in to file a reproducibility report.
Related papers
- Developmental disruption of Mef2c in Medial Ganglionic Eminence-derived cortical inhibitory interneurons impairs cellular and circuit function
Claire Ward, Kaoutsar Nasrallah, … Renata Batista‐Brito·bioRxiv·May 1, 2024
- Kif2C safeguards radial glial integrity to prevent cortical malformation
Sharmin Naher, Takako Kikkawa, … Noriko Osumi·bioRxiv·May 24, 2026
- Loss of Slc35a2 alters development of the mouse cerebral cortex
Soad Elziny, Sahibjot Sran, … Tracy A. Bedrosian·Neuroscience LettersPeer-reviewed·Jun 22, 2024
- Copb2 Is Essential For Embryogenesis And Hypomorphic Mutations Cause Human Microcephaly
Andrew DiStasio, Ashley M. Driver, … Rolf W. Stottmann·Human Molecular GeneticsPeer-reviewed·Sep 18, 2017
- MEF2C hypofunction in neuronal and neuroimmune populations cooperate to produce MEF2C haploinsufficiency syndrome-like behaviors in mice
Adam J. Harrington, Catherine Bridges, … Christopher W. Cowan·bioRxiv·Oct 30, 2019
- Mef2c Controls Postnatal Callosal Axon Targeting by Regulating Sensitivity to Ephrin Repulsion
Sriram Sudarsanam, Luis Guzman-Clavel, … Alex L. Kolodkin·Journal of NeurosciencePeer-reviewed·Apr 14, 2025