Pax6 loss alters the morphological and electrophysiological development of mouse prethalamic neurons

Development. 2022 Mar 15;149(6):dev200052. doi: 10.1242/dev.200052. Epub 2022 Mar 17.

Abstract

Pax6 is a well-known regulator of early neuroepithelial progenitor development. Its constitutive loss has a particularly strong effect on the developing prethalamus, causing it to become extremely hypoplastic. To overcome this difficulty in studying the long-term consequences of Pax6 loss for prethalamic development, we used conditional mutagenesis to delete Pax6 at the onset of neurogenesis and studied the developmental potential of the mutant prethalamic neurons in vitro. We found that Pax6 loss affected their rates of neurite elongation, the location and length of their axon initial segments, and their electrophysiological properties. Our results broaden our understanding of the long-term consequences of Pax6 deletion in the developing mouse forebrain, suggesting that it can have cell-autonomous effects on the structural and functional development of some neurons.

Keywords: Axon initial segment; Neuronal activity; Neuronal morphogenesis; Pax6; Prethalamus; Transcription factors.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Eye Proteins / metabolism
  • Gene Expression Regulation, Developmental
  • Homeodomain Proteins* / metabolism
  • Mice
  • Neurons / metabolism
  • PAX6 Transcription Factor / genetics
  • Paired Box Transcription Factors* / metabolism
  • Repressor Proteins / metabolism

Substances

  • Eye Proteins
  • Homeodomain Proteins
  • PAX6 Transcription Factor
  • Paired Box Transcription Factors
  • Pax6 protein, mouse
  • Repressor Proteins