Journal article
Prickle1 is expressed in distinct cell populations of the central nervous system and contributes to neuronal morphogenesis
Human molecular genetics, Vol.22(11), pp.2234-2246
06/01/2013
DOI: 10.1093/hmg/ddt075
PMCID: PMC3652420
PMID: 23420014
Abstract
Development of axons and dendrites constitutes a critical event in neuronal maturation and seems to require signaling through the planar cell polarity (PCP) pathway. Mutations in components of the PCP pathway lead to a spectrum of neurological phenotypes and disorders. For example, a missense mutation in
Prickle 1
(
Pk1
) is associated with progressive myoclonus epilepsy (PME) in humans, and its reduced gene dosage increases sensitivity to induced seizure in mice. In an effort to unravel the role of the PCP pathway in mammalian neuronal development, we examined the expression of
Pk1
in the central nervous system (CNS) using
in situ
hybridization (ISH) in combination with a genetic knock-in approach. We show that
Pk1
transcripts are detected in the postmitotic cells of the subplate and cortical plate during mid- and late stages of cortical neurogenesis. In adult brain,
Pk1
is expressed in distinct neuronal and glial cell populations, with dynamic formation of dendrites and glial processes during development. Of all the cell types in the mature retina, the highest expression of
Pk1
is detected in cholinergic amacrine neurons. Knockdown of
Pk1
by shRNA or dominant-negative constructs causes reduced axonal and dendritic extension in hippocampal neurons. Similarly,
Pk1
knockdown in neonatal retina leads to defects in inner and outer segments and axon terminals of photoreceptors. Our studies implicate
Pk1
function in axonal-dendritic development associated with the maturation of CNS neurons.
Details
- Title: Subtitle
- Prickle1 is expressed in distinct cell populations of the central nervous system and contributes to neuronal morphogenesis
- Creators
- Chunqiao LiuChen LinD. Thad WhitakerHirva BakeriOleg V BulgakovPinghu Liu - National Eye InstituteJingqi Lei - National Eye InstituteLijin Dong - National Eye InstituteTiansen LiAnand Swaroop
- Resource Type
- Journal article
- Publication Details
- Human molecular genetics, Vol.22(11), pp.2234-2246
- Publisher
- Oxford University Press
- DOI
- 10.1093/hmg/ddt075
- PMID
- 23420014
- PMCID
- PMC3652420
- ISSN
- 0964-6906
- eISSN
- 1460-2083
- Language
- English
- Date published
- 06/01/2013
- Academic Unit
- General Internal Medicine; Internal Medicine
- Record Identifier
- 9984094332702771
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