Cerebellar output neurons can impair non-motor behaviors by altering development of extracerebellar connectivity

Andrew S Lee1,2, Tanzil M Arefin3,4, Alina Gubanova1

  • 1Developmental Biology Program, Sloan Kettering Institute, New York, NY, USA.

Nature Communications
|February 21, 2025
PubMed

Insights

Brain compensation varies. Ablating cerebellar output neurons causes less disruption than genetic mutations (Engrailed1/2) affecting learning and memory, revealing complex cerebellar circuit interactions.

Area of Science:

  • Neuroscience
  • Developmental Biology
  • Genetics

Background:

  • Brain compensation mechanisms are crucial for development.
  • Predicting the impact of genetic mutations on neuronal function is challenging.
  • Cerebellar excitatory output neurons play a role in motor coordination and behavior.

Purpose of the Study:

  • To investigate the brain's compensatory capacity following cerebellar output neuron ablation versus genetic mutation.
  • To elucidate the distinct roles of cerebellar output neurons in motor coordination, learning, and social behaviors.
  • To understand the impact of Engrailed1/2 (EN1/2) gene loss on cerebellar function and extracerebellar circuits.

Main Methods:

  • Embryonic ablation of cerebellar excitatory output neurons in mice.
  • Genetic manipulation to induce loss of Engrailed1/2 (EN1/2) transcription factors in cerebellar excitatory neurons.
  • Behavioral testing for motor coordination, learning, and social behaviors.
  • Diffusion MRI to analyze brain connectivity.

Main Results:

  • Embryonic ablation of cerebellar output neurons allowed for significant compensation, primarily impacting motor coordination, not basic learning or social behaviors.
  • Loss of EN1/2 in cerebellar excitatory neurons resulted in deficits in adult learning and spatial working memory, despite neuronal survival.
  • Diffusion MRI revealed altered thalamo-cortico-striatal connectivity in EN1/2 mutants, indicating adverse effects on extracerebellar circuits.

Conclusions:

  • Cerebellar genetic mutations, specifically EN1/2 loss, are more disruptive to brain function than the complete absence of cerebellar output neurons.
  • The remaining cerebellar neurons lacking EN1/2 exert detrimental effects on extracerebellar circuits involved in motor learning and non-motor behaviors.
  • This study highlights the critical role of specific genes in neuronal function and brain circuit integrity, beyond mere cell presence.

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