# Molecular functions of cilia-planar polarity effectors (CPLANEs) in skin morphogenesis and homeostasis

> **NIH NIH R01** · STATE UNIVERSITY NEW YORK STONY BROOK · 2020 · $465,174

## Abstract

PROJECT SUMMARY
FUZ, INTU, and WDPCP are tissue-specific planar cell polarity (PCP) signaling effectors, and function in both
PCP and cilia formation. These PCP effectors are now collectively called cilia-planar polarity effectors
(CPLANEs). PCP and cilia perform important functions during tissue morphogenesis and homeostasis, and are
linked to neural tube defects and ciliopathies. In the mammalian skin, PCP is known to be essential for hair
patterning. The primarily cilium is essential for the hedgehog pathway – a molecular signaling mechanism
critical for hair follicle morphogenesis and the development of basal cell carcinoma. It is believed that
CPLANEs mediate the polarity cues set up by the core PCP components at the cellular level. In ciliogenesis,
CPLANEs facilitate the trafficking of intraflagellar transport proteins. However, the precise molecular
mechanism remains unclear. In this investigation, we will examine the functions of the CPLANEs in skin
morphogenesis and homeostasis. First, we will determine whether the Wdpcp gene is involved in PCP and/or
ciliogenesis in the mouse skin, and whether the CPLANE genes participate in PCP in the skin in a redundant
manner by simultaneously disrupting the Wdpcp and the Intu genes. Subsequently, we will examine the
molecular functions of the CPLANE proteins by investigating how loss-of-function and pathogenic CPLANE
mutations impair ciliogenesis. Finally, we will determine whether oncogenic mutations, responsible for
activated hedgehog signaling and basal cell carcinoma formation, could promote the formation of primary cilia
by inhibiting the cilia disassembly mechanism or by upregulating the transcription of ciliogenic genes, including
those encoding the CPLANEs. Understanding the molecular functions of CPLANEs will provide important
insight into the molecular mechanisms underlying hair follicle patterning, basal cell carcinoma formation, neural
tube defects, and ciliopathies. In the long-run, knowledge gained from this investigation will provide important
insight into how the CPLANE proteins orchestrate or segregate PCP and ciliogenesis in the mammalian skin. It
will also provide insight into how oncogenic Hh mutations reprogram the ciliogenic processes in keratinocytes,
thereby, shed light on novel mechanism of skin protection and tumor prevention.

## Key facts

- **NIH application ID:** 9852990
- **Project number:** 5R01AR061485-10
- **Recipient organization:** STATE UNIVERSITY NEW YORK STONY BROOK
- **Principal Investigator:** Jiang Chen
- **Activity code:** R01 (R01, R21, SBIR, etc.)
- **Funding institute:** NIH
- **Fiscal year:** 2020
- **Award amount:** $465,174
- **Award type:** 5
- **Project period:** 2011-07-11 → 2022-01-31

## Primary source

NIH RePORTER: https://reporter.nih.gov/project-details/9852990

## Citation

> US National Institutes of Health, RePORTER application 9852990, Molecular functions of cilia-planar polarity effectors (CPLANEs) in skin morphogenesis and homeostasis (5R01AR061485-10). Retrieved via AI Analytics 2026-05-21 from https://api.ai-analytics.org/grant/nih/9852990. Licensed CC0.

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