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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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Formato: | Texto |
Lenguaje: | English |
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Public Library of Science
2010
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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. |
format | Text |
id | pubmed-2831062 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2010 |
publisher | Public Library of Science |
record_format | MEDLINE/PubMed |
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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