Literature DB >> 29799103

c.835-5T>G Variant in SMN1 Gene Causes Transcript Exclusion of Exon 7 and Spinal Muscular Atrophy.

Shuang Wu1, Yun-Lu Li1, Ning-Yi Cheng1, Chong Wang1, En-Lin Dong1, Ying-Qian Lu1, Jin-Jing Li1, Xin-Xin Guo1, Xiang Lin1, Lu-Lu Lai1, Zhi-Wei Liu1, Ning Wang2,3, Wan-Jin Chen4,5.   

Abstract

Spinal muscular atrophy (SMA) is an autosomal recessive genetic disorder caused by survival motor neuron (SMN) protein deficiency leading the loss of motor neurons in the anterior horns of the spinal cord and brainstem. More than 95% of SMA patients are attributed to the homozygous deletion of survival motor neuron 1 (SMN1) gene, and approximately 5% are caused by compound heterozygous with a SMN1 deletion and a subtle mutation. Here, we identified a rare variant c.835-5T>G in intron 6 of SMN1 in a patient affected with type I SMA. We analyzed the functional consequences of this mutation on mRNA splicing in vitro. After transfecting pCI-SMN1, pCI-SMN2, and pCI-SMN1 c.835-5T>G minigenes into HEK293, Neuro-2a, and SHSY5Y cells, reverse transcription polymerase chain reaction (RT-PCR) was performed to compare the splicing effects of these minigenes. Finally, we found that this mutation resulted in the skipping of exon 7 in SMN1, which confirmed the genetic diagnosis of SMA.

Entities:  

Keywords:  Exon skipping; Spinal muscular atrophy; Splice variant; Survival motor neuron 1

Mesh:

Substances:

Year:  2018        PMID: 29799103     DOI: 10.1007/s12031-018-1079-1

Source DB:  PubMed          Journal:  J Mol Neurosci        ISSN: 0895-8696            Impact factor:   3.444


  19 in total

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2.  Detection of intragenic SMN1 mutations in spinal muscular atrophy patients with a single copy of SMN1.

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Journal:  J Child Neurol       Date:  2014-02-20       Impact factor: 1.987

3.  PCR-RFLP, sequencing, and quantification in molecular diagnosis of spinal muscular atrophy: limits and advantages.

Authors:  K Hamzi; H Bellayou; M Itri; S Nadifi
Journal:  J Mol Neurosci       Date:  2013-01-08       Impact factor: 3.444

4.  Quantitative analyses of SMN1 and SMN2 based on real-time lightCycler PCR: fast and highly reliable carrier testing and prediction of severity of spinal muscular atrophy.

Authors:  Markus Feldkötter; Verena Schwarzer; Radu Wirth; Thomas F Wienker; Brunhilde Wirth
Journal:  Am J Hum Genet       Date:  2001-12-21       Impact factor: 11.025

5.  Novel splice-site mutation in SMN1 associated with a very severe SMA-I phenotype.

Authors:  Dario Ronchi; Stefano Carlo Previtali; Maria Grazia Natali Sora; Graziano Barera; Benedetta Del Menico; Stefania Corti; Nereo Bresolin; Giacomo Pietro Comi
Journal:  J Mol Neurosci       Date:  2015-01-09       Impact factor: 3.444

6.  Mutation update of spinal muscular atrophy in Spain: molecular characterization of 745 unrelated patients and identification of four novel mutations in the SMN1 gene.

Authors:  Laura Alías; Sara Bernal; Pablo Fuentes-Prior; María Jesus Barceló; Eva Also; Rebeca Martínez-Hernández; Francisco J Rodríguez-Alvarez; Yolanda Martín; Elena Aller; Elena Grau; Ana Peciña; Guillermo Antiñolo; Enrique Galán; Alberto L Rosa; Miguel Fernández-Burriel; Salud Borrego; José M Millán; Concepción Hernández-Chico; Montserrat Baiget; Eduardo F Tizzano
Journal:  Hum Genet       Date:  2008-12-03       Impact factor: 4.132

7.  Antisense masking of an hnRNP A1/A2 intronic splicing silencer corrects SMN2 splicing in transgenic mice.

Authors:  Yimin Hua; Timothy A Vickers; Hazeem L Okunola; C Frank Bennett; Adrian R Krainer
Journal:  Am J Hum Genet       Date:  2008-03-27       Impact factor: 11.025

8.  The Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP INTEND): test development and reliability.

Authors:  A M Glanzman; E Mazzone; M Main; M Pelliccioni; J Wood; K J Swoboda; C Scott; M Pane; S Messina; E Bertini; E Mercuri; R S Finkel
Journal:  Neuromuscul Disord       Date:  2010-01-13       Impact factor: 4.296

9.  Natural history in proximal spinal muscular atrophy. Clinical analysis of 445 patients and suggestions for a modification of existing classifications.

Authors:  K Zerres; S Rudnik-Schöneborn
Journal:  Arch Neurol       Date:  1995-05

10.  Developmental milestones in type I spinal muscular atrophy.

Authors:  Roberto De Sanctis; Giorgia Coratti; Amy Pasternak; Jacqueline Montes; Marika Pane; Elena S Mazzone; Sally Dunaway Young; Rachel Salazar; Janet Quigley; Maria C Pera; Laura Antonaci; Leonardo Lapenta; Allan M Glanzman; Danilo Tiziano; Francesco Muntoni; Basil T Darras; Darryl C De Vivo; Richard Finkel; Eugenio Mercuri
Journal:  Neuromuscul Disord       Date:  2016-10-05       Impact factor: 4.296

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  1 in total

1.  High-throughput analysis revealed mutations' diverging effects on SMN1 exon 7 splicing.

Authors:  Přemysl Souček; Kamila Réblová; Michal Kramárek; Lenka Radová; Tereza Grymová; Pavla Hujová; Tatiana Kováčová; Matej Lexa; Lucie Grodecká; Tomáš Freiberger
Journal:  RNA Biol       Date:  2019-06-19       Impact factor: 4.652

  1 in total

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