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A novel human-specific splice isoform alters the critical C-terminus of Survival Motor Neuron protein

Spinal muscular atrophy (SMA), a leading genetic disease of children and infants, is caused by mutations or deletions of Survival Motor Neuron 1 (SMN1) gene. SMN2, a nearly identical copy of SMN1, fails to compensate for the loss of SMN1 due to skipping of exon 7. SMN2 predominantly produces SMNΔ7,...

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Autores principales: Seo, Joonbae, Singh, Natalia N., Ottesen, Eric W., Lee, Brian M., Singh, Ravindra N.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group 2016
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4969610/
https://www.ncbi.nlm.nih.gov/pubmed/27481219
http://dx.doi.org/10.1038/srep30778
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author Seo, Joonbae
Singh, Natalia N.
Ottesen, Eric W.
Lee, Brian M.
Singh, Ravindra N.
author_facet Seo, Joonbae
Singh, Natalia N.
Ottesen, Eric W.
Lee, Brian M.
Singh, Ravindra N.
author_sort Seo, Joonbae
collection PubMed
description Spinal muscular atrophy (SMA), a leading genetic disease of children and infants, is caused by mutations or deletions of Survival Motor Neuron 1 (SMN1) gene. SMN2, a nearly identical copy of SMN1, fails to compensate for the loss of SMN1 due to skipping of exon 7. SMN2 predominantly produces SMNΔ7, an unstable protein. Here we report exon 6B, a novel exon, generated by exonization of an intronic Alu-like sequence of SMN. We validate the expression of exon 6B-containing transcripts SMN6B and SMN6BΔ7 in human tissues and cell lines. We confirm generation of SMN6B transcripts from both SMN1 and SMN2. We detect expression of SMN6B protein using antibodies raised against a unique polypeptide encoded by exon 6B. We analyze RNA-Seq data to show that hnRNP C is a potential regulator of SMN6B expression and demonstrate that SMN6B is a substrate of nonsense-mediated decay. We show interaction of SMN6B with Gemin2, a critical SMN-interacting protein. We demonstrate that SMN6B is more stable than SMNΔ7 and localizes to both the nucleus and the cytoplasm. Our finding expands the diversity of transcripts generated from human SMN genes and reveals a novel protein isoform predicted to be stably expressed during conditions of stress.
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spelling pubmed-49696102016-08-11 A novel human-specific splice isoform alters the critical C-terminus of Survival Motor Neuron protein Seo, Joonbae Singh, Natalia N. Ottesen, Eric W. Lee, Brian M. Singh, Ravindra N. Sci Rep Article Spinal muscular atrophy (SMA), a leading genetic disease of children and infants, is caused by mutations or deletions of Survival Motor Neuron 1 (SMN1) gene. SMN2, a nearly identical copy of SMN1, fails to compensate for the loss of SMN1 due to skipping of exon 7. SMN2 predominantly produces SMNΔ7, an unstable protein. Here we report exon 6B, a novel exon, generated by exonization of an intronic Alu-like sequence of SMN. We validate the expression of exon 6B-containing transcripts SMN6B and SMN6BΔ7 in human tissues and cell lines. We confirm generation of SMN6B transcripts from both SMN1 and SMN2. We detect expression of SMN6B protein using antibodies raised against a unique polypeptide encoded by exon 6B. We analyze RNA-Seq data to show that hnRNP C is a potential regulator of SMN6B expression and demonstrate that SMN6B is a substrate of nonsense-mediated decay. We show interaction of SMN6B with Gemin2, a critical SMN-interacting protein. We demonstrate that SMN6B is more stable than SMNΔ7 and localizes to both the nucleus and the cytoplasm. Our finding expands the diversity of transcripts generated from human SMN genes and reveals a novel protein isoform predicted to be stably expressed during conditions of stress. Nature Publishing Group 2016-08-02 /pmc/articles/PMC4969610/ /pubmed/27481219 http://dx.doi.org/10.1038/srep30778 Text en Copyright © 2016, The Author(s) http://creativecommons.org/licenses/by/4.0/ This work is licensed under a Creative Commons Attribution 4.0 International License. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in the credit line; if the material is not included under the Creative Commons license, users will need to obtain permission from the license holder to reproduce the material. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/
spellingShingle Article
Seo, Joonbae
Singh, Natalia N.
Ottesen, Eric W.
Lee, Brian M.
Singh, Ravindra N.
A novel human-specific splice isoform alters the critical C-terminus of Survival Motor Neuron protein
title A novel human-specific splice isoform alters the critical C-terminus of Survival Motor Neuron protein
title_full A novel human-specific splice isoform alters the critical C-terminus of Survival Motor Neuron protein
title_fullStr A novel human-specific splice isoform alters the critical C-terminus of Survival Motor Neuron protein
title_full_unstemmed A novel human-specific splice isoform alters the critical C-terminus of Survival Motor Neuron protein
title_short A novel human-specific splice isoform alters the critical C-terminus of Survival Motor Neuron protein
title_sort novel human-specific splice isoform alters the critical c-terminus of survival motor neuron protein
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4969610/
https://www.ncbi.nlm.nih.gov/pubmed/27481219
http://dx.doi.org/10.1038/srep30778
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