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Endocannabinoid Enhancement via MAGL Inhibition in CDKL5 Deficiency: Selective Cellular Benefits and Domain-Specific Functional Effects in Adult <i>Cdkl5</i> KO Mice.

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Voluntary Running Improves Behavioral and Structural Abnormalities in a Mouse Model of CDKL5 Deficiency Disorder.

Nicola Mottolese1, Beatrice Uguagliati1, Marianna Tassinari1

  • 1Department of Biomedical and Neuromotor Sciences, University of Bologna, 40126 Bologna, Italy.

Biomolecules
|September 28, 2023
PubMed
Summary

Voluntary daily running improved behavioral and brain defects in a mouse model of Cyclin-dependent kinase-like 5 (CDKL5) deficiency disorder. Exercise increased neurogenesis, neuronal survival, and brain-derived neurotrophic factor (BDNF) levels.

Keywords:
Cdkl5 KO micebrain developmentdendritic pathologyneuroinflammationneuronal survivalvoluntary exercise

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Area of Science:

  • Neuroscience
  • Genetics
  • Developmental Biology

Background:

  • Cyclin-dependent kinase-like 5 (CDKL5) deficiency disorder (CDD) is a severe X-linked neurodevelopmental disease.
  • CDD presents with epilepsy, intellectual disability, and autism-like features, with no current cure.
  • The Cdkl5 knockout mouse model exhibits key CDD-related behavioral and neuronal deficits.

Purpose of the Study:

  • To investigate the therapeutic potential of voluntary exercise in the Cdkl5 knockout mouse model.
  • To determine if long-term voluntary running can ameliorate behavioral and neurobiological deficits in CDD.

Main Methods:

  • Cdkl5 knockout mice underwent a month of voluntary daily running.
  • Behavioral tests assessed locomotion, impulsivity, and memory.
  • Brain tissue analysis examined neurogenesis, neuronal survival, dendritic spine maturation, microglia activation, and BDNF levels.

Main Results:

  • Voluntary running significantly improved hyperlocomotion, impulsivity, and memory performance in Cdkl5 KO mice.
  • Exercise led to increased hippocampal neurogenesis, neuronal survival, and spine maturation.
  • Running inhibited microglia activation and elevated BDNF levels in the brain.

Conclusions:

  • Long-term voluntary exercise demonstrates significant benefits for the CDD mouse model.
  • Exercise positively impacts behavioral and structural brain deficits associated with CDKL5 deficiency.
  • Exercise, potentially via BDNF, may serve as an effective adjuvant therapy for CDD.