Mtor/Rptor Function Globally Prevents Cortical Microcephaly and Cell-autonomously Promotes Postnatal Neuron Survival in Cell Type Specific Manner
Ana VillalbaiD, Robert Beattie, Florian M. PauleriD, Carmen Streicher, Osvaldo A. MirandaiD, Thomas KrausgruberiD, Martin SenekowitschiD, Matthias FarlikiD, Christoph BockiD, Thomas Ruelicke, Simon HippenmeyeriD
Abstract
The generation of faithful cell-type diversity and correct projection neuron numbers is essential for cerebral cortex development. Corticogenesis is however susceptible to genetic interference of critical signaling pathways, including mutations in Mtor/Rptor that lead to microcephaly. How the loss of Rptor/mTORC1 function affects cortical developmental programs, at single cell level, is still unknown. Here, we utilized Mosaic Analysis with Double Markers (MADM) technology to probe Rptor gene function upon sparse single cell- or global tissue-wide ablation. We found that tissue-wide effects drive the etiology of cortical microcephaly upon loss of Rptor, rather than deficits in projection neuron genesis. Conversely, Rptor function is cell-autonomously required for postnatal projection neuron survival in a highly cell-type-specific manner. Collectively, our results suggest that the fine balance of precise cell-type-specific cell-autonomous Rptor/mTORC1 function in concert with non-cell-autonomous tissue-wide effects is essential for the development of a properly-sized cerebral cortex with accurate projection neuron diversity.
02 /Specification & Classification
Click any tag to browse matching papersReproducibility 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
- Tissue-wide Effects Overrule Cell-intrinsic Gene Function in Cortical Projection Neuron Migration
Andi H. Hansen, Florian M. Pauler, … Simon Hippenmeyer·bioRxiv·Feb 16, 2022
- The mTOR pathway genes mTOR, Rheb, Depdc5, Pten, and Tsc1 have convergent and divergent impacts on cortical neuron development and function
Lena H. Nguyen, Youfen Xu, … Angélique Bordey·bioRxiv·Aug 11, 2023
- mTOR signaling regulates the morphology and migration of outer radial glia in developing human cortex
Madeline G. Andrews, Lakshmi Subramanian, Arnold R. Kriegstein·eLifePeer-reviewed·Sep 2, 2020
- Pten Orchestrates Neurogenic Radial Glia Lineage Progression and Tunes Neocortical Astrocyte Production
Osvaldo A. Miranda, Ximena Contreras, … Simon Hippenmeyer·bioRxiv·May 5, 2026
- A microRNA cluster downstream of the selector gene Fezf2 coordinates fate specification with dendritic branching in cortical neurons
Asha Iyer, Verl B. Siththanandan, … Suzanne A. Tharin·bioRxiv·Nov 3, 2021
- mTOR driven gene transcription is required for cholesterol production in neurons of the developing cerebral cortex
Martin Schüle, Tamer Butto, … Jennifer Winter·bioRxiv·Sep 4, 2020