Literature DB >> 26724723

Spinal Muscular Atrophy: Overview of Molecular Diagnostic Approaches.

Thomas W Prior1, Narasimhan Nagan2.   

Abstract

Spinal muscular atrophy (SMA) is an autosomal recessive neurodegenerative disease and the most common genetic cause of infant mortality, affecting ∼1 in 10,000 live births. The disease is characterized by progressive symmetrical muscle weakness resulting from the degeneration and loss of anterior horn cells in the spinal cord and brain stem nuclei. The disease is classified on the basis of age of onset and clinical course. SMA is caused by mutations in the telomeric copy of the survival motor neuron 1 (SMN1) gene, but all patients retain a centromeric copy of the gene, SMN2. The homozygous absence of the SMN1 exon 7 has been observed in the majority of patients and is being utilized as a reliable and sensitive SMA diagnostic test. In the majority of cases, the disease severity correlates inversely with an increased SMN2 gene copy number. Carrier detection, in the deletion cases, relies on the accurate determination of the SMN1 gene copies. Since SMA is one of the most common lethal genetic disorders, with a carrier frequency of 1 in 40 to 1 in 60, direct carrier dosage testing has been beneficial to many families. This unit attempts to highlight the molecular genetics of SMA with a focus on the advantages and limitations of the current molecular technologies.
Copyright © 2016 John Wiley & Sons, Inc.

Entities:  

Keywords:  SMN1; SMN2; carrier testing; genetic testing; spinal muscular atrophy; survival motor neuron

Mesh:

Substances:

Year:  2016        PMID: 26724723     DOI: 10.1002/0471142905.hg0927s88

Source DB:  PubMed          Journal:  Curr Protoc Hum Genet        ISSN: 1934-8258


  5 in total

1.  Conditional deletion of SMN in cell culture identifies functional SMN alleles.

Authors:  Anton J Blatnik; Vicki L McGovern; Thanh T Le; Chitra C Iyer; Brian K Kaspar; Arthur H M Burghes
Journal:  Hum Mol Genet       Date:  2020-10-19       Impact factor: 6.150

2.  Mild SMN missense alleles are only functional in the presence of SMN2 in mammals.

Authors:  Chitra C Iyer; Kaitlyn M Corlett; Aurélie Massoni-Laporte; Sandra I Duque; Narasimhan Madabusi; Sarah Tisdale; Vicki L McGovern; Thanh T Le; Phillip G Zaworski; W David Arnold; Livio Pellizzoni; Arthur H M Burghes
Journal:  Hum Mol Genet       Date:  2018-10-01       Impact factor: 6.150

3.  Accurate diagnosis of spinal muscular atrophy and 22q11.2 deletion syndrome using limited deoxynucleotide triphosphates and high-resolution melting.

Authors:  Xiaoqing Zhang; Bo Wang; Lichen Zhang; Guoling You; Robert A Palais; Luming Zhou; Qihua Fu
Journal:  BMC Genomics       Date:  2018-06-20       Impact factor: 3.969

4.  Newborn screening for spinal muscular atrophy: The views of affected families and adults.

Authors:  Felicity K Boardman; Philip J Young; Frances E Griffiths
Journal:  Am J Med Genet A       Date:  2017-04-04       Impact factor: 2.802

5.  A 20-year Clinical and Genetic Neuromuscular Cohort Analysis in Lebanon: An International Effort.

Authors:  Andre Megarbane; Sami Bizzari; Asha Deepthi; Sandra Sabbagh; Hicham Mansour; Eliane Chouery; Ghassan Hmaimess; Rosette Jabbour; Cybel Mehawej; Saada Alame; Abeer Hani; Dana Hasbini; Ismat Ghanem; Salam Koussa; Mahmoud Taleb Al-Ali; Marc Obeid; Diana Bou Talea; Gerard Lefranc; Nicolas Lévy; France Leturcq; Stephany El Hayek; Valérie Delague; J Andoni Urtizberea
Journal:  J Neuromuscul Dis       Date:  2022
  5 in total

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