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Role of Spastin in Apical Domain Control along the Rhabdomere Elongation in Drosophila Photoreceptor

BACKGROUND: Mutations in spastin are the most common cause of hereditary spastin paraplegia, a neurodegenerative disease. In this study, the role of spastin was examined in Drosophila photoreceptor development. METHODOLOGY/PRINCIPAL FINDINGS: The spastin mutation in developing pupal eyes causes a mi...

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Autores principales: Chen, Geng, League, Garrett P., Nam, Sang-Chul
Formato: Texto
Lenguaje:English
Publicado: Public Library of Science 2010
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2831062/
https://www.ncbi.nlm.nih.gov/pubmed/20209135
http://dx.doi.org/10.1371/journal.pone.0009480
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author Chen, Geng
League, Garrett P.
Nam, Sang-Chul
author_facet Chen, Geng
League, Garrett P.
Nam, Sang-Chul
author_sort Chen, Geng
collection PubMed
description BACKGROUND: Mutations in spastin are the most common cause of hereditary spastin paraplegia, a neurodegenerative disease. In this study, the role of spastin was examined in Drosophila photoreceptor development. METHODOLOGY/PRINCIPAL FINDINGS: The spastin mutation in developing pupal eyes causes a mild mislocalization of the apical membrane domain at the distal section, but the apical domain was dramatically reduced at the proximal section of the developing pupal eye. Since the rhabdomeres in developing pupal eyes grow from distal to proximal, this phenotype strongly suggests that spastin is required for apical domain maintenance during rhabdomere elongation. This role of spastin in apical domain modulation was further supported by spastin's gain-of-function phenotype. Spastin overexpression in photoreceptors caused the expansion of the apical membrane domain from apical to basolateral in the developing photoreceptor. Although the localizations of the apical domain and adherens junctions (AJs) were severely expanded, there were no defects in cell polarity. CONCLUSIONS/SIGNIFICANCE: These results strongly suggest that spastin is essential for apical domain biogenesis during rhabdomere elongation in Drosophila photoreceptor morphogenesis.
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spelling pubmed-28310622010-03-06 Role of Spastin in Apical Domain Control along the Rhabdomere Elongation in Drosophila Photoreceptor Chen, Geng League, Garrett P. Nam, Sang-Chul PLoS One Research Article BACKGROUND: Mutations in spastin are the most common cause of hereditary spastin paraplegia, a neurodegenerative disease. In this study, the role of spastin was examined in Drosophila photoreceptor development. METHODOLOGY/PRINCIPAL FINDINGS: The spastin mutation in developing pupal eyes causes a mild mislocalization of the apical membrane domain at the distal section, but the apical domain was dramatically reduced at the proximal section of the developing pupal eye. Since the rhabdomeres in developing pupal eyes grow from distal to proximal, this phenotype strongly suggests that spastin is required for apical domain maintenance during rhabdomere elongation. This role of spastin in apical domain modulation was further supported by spastin's gain-of-function phenotype. Spastin overexpression in photoreceptors caused the expansion of the apical membrane domain from apical to basolateral in the developing photoreceptor. Although the localizations of the apical domain and adherens junctions (AJs) were severely expanded, there were no defects in cell polarity. CONCLUSIONS/SIGNIFICANCE: These results strongly suggest that spastin is essential for apical domain biogenesis during rhabdomere elongation in Drosophila photoreceptor morphogenesis. Public Library of Science 2010-03-03 /pmc/articles/PMC2831062/ /pubmed/20209135 http://dx.doi.org/10.1371/journal.pone.0009480 Text en Chen et al. http://creativecommons.org/licenses/by/4.0/ This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are properly credited.
spellingShingle Research Article
Chen, Geng
League, Garrett P.
Nam, Sang-Chul
Role of Spastin in Apical Domain Control along the Rhabdomere Elongation in Drosophila Photoreceptor
title Role of Spastin in Apical Domain Control along the Rhabdomere Elongation in Drosophila Photoreceptor
title_full Role of Spastin in Apical Domain Control along the Rhabdomere Elongation in Drosophila Photoreceptor
title_fullStr Role of Spastin in Apical Domain Control along the Rhabdomere Elongation in Drosophila Photoreceptor
title_full_unstemmed Role of Spastin in Apical Domain Control along the Rhabdomere Elongation in Drosophila Photoreceptor
title_short Role of Spastin in Apical Domain Control along the Rhabdomere Elongation in Drosophila Photoreceptor
title_sort role of spastin in apical domain control along the rhabdomere elongation in drosophila photoreceptor
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2831062/
https://www.ncbi.nlm.nih.gov/pubmed/20209135
http://dx.doi.org/10.1371/journal.pone.0009480
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