Ataxin-2 is essential for cytoskeletal dynamics and neurodevelopment in Drosophila

Urko del Castillo*, Rosalind Norkett, Wen Lu, Anna Serpinskaya, Vladimir I. Gelfand

*Corresponding author for this work

Research output: Contribution to journalArticlepeer-review

1 Scopus citations


Ataxin-2 (Atx2) is a highly conserved RNA binding protein. Atx2 undergoes polyglutamine expansion leading to amyotrophic lateral sclerosis (ALS) or spinocerebellar ataxia type 2 (SCA2). However, the physiological functions of Atx2 in neurons remain unknown. Here, using the powerful genetics of Drosophila, we show that Atx2 is essential for normal neuronal cytoskeletal dynamics and organelle trafficking. Upon neuron-specific Atx2 loss, the microtubule and actin networks were abnormally stabilized and cargo transport was drastically inhibited. Depletion of Atx2 caused multiple morphological defects in the nervous system of third instar larvae. These include reduced brain size, impaired axon development, and decreased dendrite outgrowth. Defects in the nervous system caused loss of the ability to crawl and lethality at the pupal stage. Taken together, these data mark Atx2 as a major regulator of cytoskeletal dynamics and denote Atx2 as an essential gene in neurodevelopment, as well as a neurodegenerative factor.

Original languageEnglish (US)
Article number103536
Issue number1
StatePublished - Jan 21 2022


  • Biological sciences
  • Cell biology
  • Cellular neuroscience
  • Developmental biology
  • Developmental neuroscience
  • Neuroscience

ASJC Scopus subject areas

  • General


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