Jove
Visualize
Contact Us
JoVE
x logofacebook logolinkedin logoyoutube logo
ABOUT JoVE
OverviewLeadershipBlogJoVE Help Center
AUTHORS
Publishing ProcessEditorial BoardScope & PoliciesPeer ReviewFAQSubmit
LIBRARIANS
TestimonialsSubscriptionsAccessResourcesLibrary Advisory BoardFAQ
RESEARCH
JoVE JournalMethods CollectionsJoVE Encyclopedia of ExperimentsArchive
EDUCATION
JoVE CoreJoVE BusinessJoVE Science EducationJoVE Lab ManualFaculty Resource CenterFaculty Site
Terms & Conditions of Use
Privacy Policy
Policies

Related Concept Videos

Neurulation01:30

Neurulation

Neurulation is the embryological process which forms the precursors of the central nervous system and occurs after gastrulation has established the three primary cell layers of the embryo: ectoderm, mesoderm, and endoderm. In humans, the majority of this system is formed via primary neurulation, in which the central portion of the ectoderm—originally appearing as a flat sheet of cells—folds upwards and inwards, sealing off to form a hollow neural tube. As development proceeds, the anterior...

You might also read

Related Articles

Articles linked to this work by shared authors, journal, and citation graph.

Sort by
Same author

Loss of INPP5E affects photoreceptor outer segment membrane biogenesis in iPSC-derived human retinal organoids.

Journal of cell science·2026
Same author

Identifiability and Model Misspecification for Modelling Recurrent Infections Using Routine Health Care Data.

American journal of epidemiology·2026
Same author

Autism-like phenotypes and increased NMDAR2D expression in mice with KDM5B histone lysine demethylase deficiency.

Science advances·2026
Same author

Autism subtypes identified using cross-species functional connectivity analyses.

Nature neuroscience·2026
Same author

Impact of grassroots development of interprofessional team-based practices: Retrospective matched cohort study using ICES data.

Canadian family physician Medecin de famille canadien·2026
Same author

Apples and oranges: Better harmonisation of vaccine trials is needed to inform inclusion in immunisation programs.

Vaccine·2026

Related Experiment Video

Updated: May 18, 2026

Stem cell-like Xenopus Embryonic Explants to Study Early Neural Developmental Features In Vitro and In Vivo
11:13

Stem cell-like Xenopus Embryonic Explants to Study Early Neural Developmental Features In Vitro and In Vivo

Published on: February 2, 2016

Gli3 controls corpus callosum formation by positioning midline guideposts during telencephalic patterning.

Dario Magnani1, Kerstin Hasenpusch-Theil, Carine Benadiba

  • 1Centre for Integrative Physiology, University of Edinburgh, Edinburgh, UK.

Cerebral Cortex (New York, N.Y. : 1991)
|October 9, 2012
PubMed
Summary

Gli3 gene mutations disrupt midline guidepost positioning, causing agenesis of the corpus callosum (CC). Correcting Slit2 expression partially restores CC development, highlighting Gli3

Keywords:
Fgf8Gli3PdnSlit2corpus callosum

More Related Videos

Dissection and Lateral Mounting of Zebrafish Embryos: Analysis of Spinal Cord Development
05:36

Dissection and Lateral Mounting of Zebrafish Embryos: Analysis of Spinal Cord Development

Published on: February 28, 2014

Real-time Bioluminescence Imaging of Notch Signaling Dynamics during Murine Neurogenesis
10:25

Real-time Bioluminescence Imaging of Notch Signaling Dynamics during Murine Neurogenesis

Published on: December 12, 2019

Related Experiment Videos

Last Updated: May 18, 2026

Stem cell-like Xenopus Embryonic Explants to Study Early Neural Developmental Features In Vitro and In Vivo
11:13

Stem cell-like Xenopus Embryonic Explants to Study Early Neural Developmental Features In Vitro and In Vivo

Published on: February 2, 2016

Dissection and Lateral Mounting of Zebrafish Embryos: Analysis of Spinal Cord Development
05:36

Dissection and Lateral Mounting of Zebrafish Embryos: Analysis of Spinal Cord Development

Published on: February 28, 2014

Real-time Bioluminescence Imaging of Notch Signaling Dynamics during Murine Neurogenesis
10:25

Real-time Bioluminescence Imaging of Notch Signaling Dynamics during Murine Neurogenesis

Published on: December 12, 2019

Area of Science:

  • Neuroscience
  • Developmental Biology
  • Genetics

Background:

  • The corpus callosum (CC) is essential for interhemispheric communication, formed by axons crossing the brain midline.
  • Glial and neuronal guideposts are critical for directing callosal axon pathfinding, but their precise positioning mechanisms are unclear.
  • The Gli3 gene plays a role in forebrain development, but its specific function in CC formation is not fully understood.

Purpose of the Study:

  • To investigate the role of the Gli3 gene in the development and positioning of midline guidepost cells.
  • To elucidate the molecular mechanisms underlying corpus callosum agenesis in Gli3 mutant mice.
  • To determine the contribution of Slit2, Fgf, and Wnt/β-catenin signaling pathways to CC development.

Main Methods:

  • Analysis of the Gli3 hypomorphic mouse mutant, Polydactyly Nagoya (Pdn), exhibiting CC agenesis.
  • Transplantation experiments to assess the causality of midline defects in CC agenesis.
  • Gene expression analysis (Slit2) and manipulation of signaling pathways (Fgf, Wnt/β-catenin) in mouse embryos.
  • Genetic rescue experiments using compound mutants (Pdn/Pdn;Slit2-/-).

Main Results:

  • Gli3 mutant (Pdn) mice display agenesis of the corpus callosum and misplacement of midline guidepost cells.
  • CC agenesis is primarily caused by midline defects originating during telencephalic patterning.
  • Gli3 mutations lead to altered Slit2 expression and dysregulated Fgf and Wnt/β-catenin signaling.
  • Mimicking these signaling changes with sprouty1/2 mutations also results in guidepost disorganization and CC agenesis.
  • Partial rescue of midline abnormalities in Pdn/Pdn;Slit2(-/-) embryos confirms Slit2's critical role.

Conclusions:

  • Gli3 is crucial for restricting Slit2 expression and modulating Fgf and Wnt/β-catenin signaling pathways.
  • Proper regulation of these signaling pathways by Gli3 is essential for the correct positioning of midline guideposts.
  • Dysregulation of Gli3-controlled signaling disrupts guidepost formation, leading to corpus callosum agenesis and impaired brain development.