Tst-1/Oct-6/SCIP regulates a unique step in peripheral myelination and is required for normal respiration

John R. Bermingham, Steven S. Scherer, Shawn O'Connell, Edgardo Arroyo, Kristin A. Kalla, Frank L. Powell, Michael G. Rosenfeld

Research output: Contribution to journalArticlepeer-review

223 Scopus citations

Abstract

The terminal differentiation of myelinating glia involves complex interactions that culminate in the formation of myelin. The POU domain transcription factor Tst-1/Oct-6/SCIP is expressed transiently during myelination, and we report here that it has a critical role in this developmental process. Deletion of the Tst-1/Oct-6/SCIP gene produces a severe defect in peripheral myelination by arresting Schwann cell maturation before axonal wrapping. Unexpectedly, the activation of major myelin-specific genes appears to be unaffected by the Tst-1/Oct-6/SCIP mutation, demonstrating that multiple, independently regulated events are required for terminal differentiation of Schwann cells. In addition, aberrant differentiation and migration of specific neurons in Tst-1/Oct-6/SCIP mutant homozygotes is associated with a fatal breathing defect, providing a model for investigating the regulation of pulmonary homeostasis.

Original languageEnglish
Pages (from-to)1751-1762
Number of pages12
JournalGenes and Development
Volume10
Issue number14
DOIs
StatePublished - 1996

Keywords

  • Oct-6
  • POU domain transcription factor
  • SCIP
  • Schwann cell
  • Tst- 1
  • neuronal migration
  • nucleus of the lateral olfactory tract
  • peripheral myelination
  • phrenic nucleus
  • respiration

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