Region-specific reversal of epidermal planar polarity in the rosette fancy mouse

Maureen Cetera, Rishabh Sharan, Gabriela Hayward-Lara, Brooke Phillips, Abhishek Biswas, Madalene Halley, Evalyn Beall, Bridgett vonHoldt, Danelle Devenport

Research output: Contribution to journalArticlepeer-review

1 Scopus citations

Abstract

The planar cell polarity (PCP) pathway collectively orients cells with respect to a body axis. Hair follicles of the murine epidermis provide a striking readout of PCP activity in their uniform alignment across the skin. Here, we characterize, from the molecular to tissue-scale, PCP establishment in the rosette fancy mouse, a natural variant with posterior-specific whorls in its fur, to understand how epidermal polarity is coordinated across the tissue. We find that rosette hair follicles emerge with reversed orientations specifically in the posterior region, creating a mirror image of epidermal polarity. The rosette trait is associated with a missense mutation in the core PCP gene Fzd6, which alters a consensus site for N-linked glycosylation, inhibiting its membrane localization. Unexpectedly, the Fzd6 trafficking defect does not block asymmetric localization of the other PCP proteins. Rather, the normally uniform axis of PCP asymmetry rotates where the PCP-directed cell movements that orient follicles are reversed, suggesting the PCP axis rotates 180°. Collectively, our multiscale analysis of epidermal polarity reveals PCP patterning can be regionally decoupled to produce posterior whorls in the rosette fancy mouse.

Original languageEnglish (US)
Article numberdev202078
JournalDevelopment (Cambridge)
Volume150
Issue number17
DOIs
StatePublished - Sep 2023

Bibliographical note

Publisher Copyright:
© 2023. Published by The Company of Biologists Ltd.

Keywords

  • Epidermis
  • Frizzled 6
  • Hair follicle
  • Morphogenesis
  • Mouse
  • Planar cell polarity

PubMed: MeSH publication types

  • Journal Article
  • Research Support, N.I.H., Extramural
  • Research Support, U.S. Gov't, Non-P.H.S.

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