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JMJD-1.2/PHF8 controls axon guidance by regulating Hedgehog-like signaling
Riveiro, Alba Redo1,2,5; Mariani, Luca1,2,5; Malmberg, Emily1,2; Amendola, Pier Giorgio1,2; Peltonen, Juhani3; Wong, Garry4; Salcini, Anna Elisabetta1,2
2017-03
Source PublicationDEVELOPMENT
ISSN0950-1991
Volume144Issue:5Pages:856-865
Abstract

Components of the KDM7 family of histone demethylases are implicated in neuronal development and one member, PHF8, is often found to be mutated in cases of X-linked mental retardation. However, how PHF8 regulates neurodevelopmental processes and contributes to the disease is still largely unknown. Here, we show that the catalytic activity of a PHF8 homolog in Caenorhabditis elegans, JMJD-1.2, is required non-cell-autonomously for proper axon guidance. Loss of JMJD-1.2 dysregulates transcription of the Hedgehog-related genes wrt-8 and grl-16, the overexpression of which is sufficient to induce the axonal defects. Deficiency of either wrt-8 or grl-16, or reduced expression of homologs of genes promoting Hedgehog signaling, restores correct axon guidance in jmjd-1.2 mutants. Genetic and overexpression data indicate that Hedgehog-related genes act on axon guidance through actin remodelers. Thus, our study highlights a novel function of jmjd-1.2 in axon guidance that might be relevant for the onset of X-linked mental retardation and provides compelling evidence of a conserved function of the Hedgehog pathway in C. elegans axon migration.

KeywordEpigenetics Histone Demethylase Neuronal Development Axon Guidance Hedgehog Signaling C. Elegans
DOIhttp://doi.org/10.1242/dev.142695
URLView the original
Indexed BySCI
Language英语
WOS Research AreaDevelopmental Biology
WOS SubjectDevelopmental Biology
WOS IDWOS:000395650100013
PublisherCOMPANY OF BIOLOGISTS LTD
The Source to ArticleWOS
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Cited Times [WOS]:5   [WOS Record]     [Related Records in WOS]
Document TypeJournal article
CollectionFaculty of Health Sciences
Corresponding AuthorSalcini, Anna Elisabetta
Affiliation1.Biotech Research & Innovation Centre (BRIC), University of Copenhagen, 2200, Copenhagen, Denmark
2.Centre for Epigenetics, University of Copenhagen, 2200, Copenhagen, Denmark
3.A. I. Virtanen Institute for Molecular Sciences, Department of Neurobiology, University of Eastern Finland, 70211, Kuopio, Finland
4.Faculty of Health Sciences, University of Macau, 999078, Macau, China
5.The Danish Stem Cell Center (DanStem), University of Copenhagen, 2200, Copenhagen, Denmark
Recommended Citation
GB/T 7714
Riveiro, Alba Redo,Mariani, Luca,Malmberg, Emily,et al. JMJD-1.2/PHF8 controls axon guidance by regulating Hedgehog-like signaling[J]. DEVELOPMENT,2017,144(5):856-865.
APA Riveiro, Alba Redo.,Mariani, Luca.,Malmberg, Emily.,Amendola, Pier Giorgio.,Peltonen, Juhani.,...&Salcini, Anna Elisabetta.(2017).JMJD-1.2/PHF8 controls axon guidance by regulating Hedgehog-like signaling.DEVELOPMENT,144(5),856-865.
MLA Riveiro, Alba Redo,et al."JMJD-1.2/PHF8 controls axon guidance by regulating Hedgehog-like signaling".DEVELOPMENT 144.5(2017):856-865.
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