Cargando…
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,...
Autores principales: | , , , , |
---|---|
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 |
_version_ | 1782445807288451072 |
---|---|
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. |
format | Online Article Text |
id | pubmed-4969610 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2016 |
publisher | Nature Publishing Group |
record_format | MEDLINE/PubMed |
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 |
work_keys_str_mv | AT seojoonbae anovelhumanspecificspliceisoformaltersthecriticalcterminusofsurvivalmotorneuronprotein AT singhnatalian anovelhumanspecificspliceisoformaltersthecriticalcterminusofsurvivalmotorneuronprotein AT ottesenericw anovelhumanspecificspliceisoformaltersthecriticalcterminusofsurvivalmotorneuronprotein AT leebrianm anovelhumanspecificspliceisoformaltersthecriticalcterminusofsurvivalmotorneuronprotein AT singhravindran anovelhumanspecificspliceisoformaltersthecriticalcterminusofsurvivalmotorneuronprotein AT seojoonbae novelhumanspecificspliceisoformaltersthecriticalcterminusofsurvivalmotorneuronprotein AT singhnatalian novelhumanspecificspliceisoformaltersthecriticalcterminusofsurvivalmotorneuronprotein AT ottesenericw novelhumanspecificspliceisoformaltersthecriticalcterminusofsurvivalmotorneuronprotein AT leebrianm novelhumanspecificspliceisoformaltersthecriticalcterminusofsurvivalmotorneuronprotein AT singhravindran novelhumanspecificspliceisoformaltersthecriticalcterminusofsurvivalmotorneuronprotein |