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Related Experiment Video

Updated: Apr 19, 2026

Whole-mount Imaging of Mouse Embryo Sensory Axon Projections
08:37

Whole-mount Imaging of Mouse Embryo Sensory Axon Projections

Published on: December 9, 2014

10.5K

LIS1 Is Critical for Axon Integrity in Adult Mice.

Samaneh Matoo1, Anne M Ventrone1, Shreena Patel1

  • 1Department of Biological Sciences, University of South Carolina, Columbia, South Carolina 29208.

The Journal of Neuroscience : the Official Journal of the Society for Neuroscience
|April 17, 2026
PubMed
Summary

LIS1 depletion in adult mice causes motor deficits and lethality by damaging projection neuron axons. This LIS1 (LIS1) protein regulates dynein (dynein), a motor protein, suggesting potential therapeutic targets for neurodegenerative diseases.

Keywords:
LIS1PAFAH1B1adult mouse astrocyteadult mouse neuroncytoplasmic dyneindegeneration

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Area of Science:

  • Neuroscience
  • Cell Biology
  • Genetics

Background:

  • LIS1 mutations cause lissencephaly, a developmental brain malformation.
  • LIS1 is also expressed in adult mouse tissues, but its role in mature neurons is less understood.

Purpose of the Study:

  • To investigate the function of LIS1 in adult projection neurons and its contribution to the lethal phenotype observed in LIS1 knockout mice.
  • To determine if LIS1 depletion in specific cell types underlies the observed nervous system dysfunction.

Main Methods:

  • Inducible LIS1 knockout (iKO) in adult mice using a Cre-Lox system.
  • Selective depletion of LIS1 in astrocytes and projection neurons.
  • Assessment of motor function, survival rates, and axonal integrity.
  • In vitro culture of sensory neurons from iKO mice.

Main Results:

  • LIS1 depletion from astrocytes did not cause lethality.
  • LIS1 depletion from projection neurons led to motor deficits and rapid lethality in both sexes.
  • Progressive, widespread axonal degeneration was observed in both motor and sensory axons.
  • Cultured sensory neurons from iKO mice exhibited axonal swellings and fragmentation.

Conclusions:

  • LIS1 is essential for maintaining axonal integrity in mature projection neurons.
  • LIS1 depletion disrupts dynein function, leading to Wallerian-like axon degeneration.
  • Targeting Wallerian degeneration may offer therapeutic strategies for dynein-related neurodegenerative diseases.