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Tissue specific roles for the Ribosome Biogenesis Factor Wdr43 in zebrafish development

Zhao, Chengtian, Andreeva, Viktoria, Gibert, Yann, LaBonty, Melissa, Lattanzi, Victoria, Prabhudesai, Shubhangi, Zhou, Yi, Zon, Leonard, McCann, Kathleen L., Baserga, Susan and Yelick, Pamela C. 2014, Tissue specific roles for the Ribosome Biogenesis Factor Wdr43 in zebrafish development, PLoS genetics, vol. 10, no. 1, pp. 1-15, doi: 10.1371/journal.pgen.1004074.

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Title Tissue specific roles for the Ribosome Biogenesis Factor Wdr43 in zebrafish development
Author(s) Zhao, Chengtian
Andreeva, Viktoria
Gibert, Yann
LaBonty, Melissa
Lattanzi, Victoria
Prabhudesai, Shubhangi
Zhou, Yi
Zon, Leonard
McCann, Kathleen L.
Baserga, Susan
Yelick, Pamela C.
Journal name PLoS genetics
Volume number 10
Issue number 1
Article ID e1004074
Start page 1
End page 15
Total pages 15
Publisher Public Library of Science
Place of publication San Francisco, Calif.
Publication date 2014
ISSN 1553-7390
Summary During vertebrate craniofacial development, neural crest cells (NCCs) contribute to most of the craniofacial pharyngeal skeleton. Defects in NCC specification, migration and differentiation resulting in malformations in the craniofacial complex are associated with human craniofacial disorders including Treacher-Collins Syndrome, caused by mutations in TCOF1. It has been hypothesized that perturbed ribosome biogenesis and resulting p53 mediated neuroepithelial apoptosis results in NCC hypoplasia in mouse Tcof1 mutants. However, the underlying mechanisms linking ribosome biogenesis and NCC development remain poorly understood. Here we report a new zebrafish mutant, fantome (fan), which harbors a point mutation and predicted premature stop codon in zebrafish wdr43, the ortholog to yeast UTP5. Although wdr43 mRNA is widely expressed during early zebrafish development, and its deficiency triggers early neural, eye, heart and pharyngeal arch defects, later defects appear fairly restricted to NCC derived craniofacial cartilages. Here we show that the C-terminus of Wdr43, which is absent in fan mutant protein, is both necessary and sufficient to mediate its nucleolar localization and protein interactions in metazoans. We demonstrate that Wdr43 functions in ribosome biogenesis, and that defects observed in fan mutants are mediated by a p53 dependent pathway. Finally, we show that proper localization of a variety of nucleolar proteins, including TCOF1, is dependent on that of WDR43. Together, our findings provide new insight into roles for Wdr43 in development, ribosome biogenesis, and also ribosomopathy-induced craniofacial phenotypes including Treacher-Collins Syndrome.
Language eng
DOI 10.1371/journal.pgen.1004074
Field of Research 119999 Medical and Health Sciences not elsewhere classified
Socio Economic Objective 970111 Expanding Knowledge in the Medical and Health Sciences
HERDC Research category C1.1 Refereed article in a scholarly journal
Copyright notice ©2014, The Authors
Free to Read? Yes
Use Rights Creative Commons Attribution licence
Persistent URL http://hdl.handle.net/10536/DRO/DU:30063546

Document type: Journal Article
Collections: School of Medicine
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Created: Fri, 23 May 2014, 09:41:19 EST

Every reasonable effort has been made to ensure that permission has been obtained for items included in DRO. If you believe that your rights have been infringed by this repository, please contact drosupport@deakin.edu.au.