Literature DB >> 28797588

Longitudinal assessments in discordant twins with SMA.

Marika Pane1, Leonardo Lapenta1, Emanuela Abiusi2, Roberto de Sanctis3, Marco Luigetti4, Concetta Palermo3, Domiziana Ranalli1, Stefania Fiori2, Francesco Danilo Tiziano2, Eugenio Mercuri5.   

Abstract

We report longitudinal clinical and neurophysiological assessments in twins affected by spinal muscular atrophy (SMA) with discordant phenotypes. The boy had the homozygous deletion of SMN1, a typical type 1 SMA course, and died at the age of eight months. His twin sister, asymptomatic at the time of the diagnosis in her brother, had the same genetic defect but she developed clinical and electrophysiological signs of type 2 SMA. The reduction of tendon reflexes was the first clinical sign at the age of 4 months, followed within few weeks, by a mild decrement in the amplitude of the compound motor action potentials. After the age of 9 months, she showed a sudden clinical and electrophysiological deterioration. Among molecular tests, we determined SMN2 copy number, SMN2 and Plastin 3 transcript levels in peripheral blood, and observed no relevant differences between twins.
Copyright © 2017. Published by Elsevier B.V.

Entities:  

Keywords:  CMAP; Modifiers; Phenotypes; Spinal muscular atrophy; Twins

Mesh:

Substances:

Year:  2017        PMID: 28797588     DOI: 10.1016/j.nmd.2017.06.559

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  5 in total

1.  Complete sequencing of the SMN2 gene in SMA patients detects SMN gene deletion junctions and variants in SMN2 that modify the SMA phenotype.

Authors:  Corey Ruhno; Vicki L McGovern; Matthew R Avenarius; Pamela J Snyder; Thomas W Prior; Flavia C Nery; Abdurrahman Muhtaseb; Jennifer S Roggenbuck; John T Kissel; Valeria A Sansone; Jennifer J Siranosian; Alec J Johnstone; Pann H Nwe; Ren Z Zhang; Kathryn J Swoboda; Arthur H M Burghes
Journal:  Hum Genet       Date:  2019-02-20       Impact factor: 4.132

Review 2.  Spinal muscular atrophy - insights and challenges in the treatment era.

Authors:  Eugenio Mercuri; Maria Carmela Pera; Mariacristina Scoto; Richard Finkel; Francesco Muntoni
Journal:  Nat Rev Neurol       Date:  2020-10-14       Impact factor: 42.937

Review 3.  Spinal muscular atrophy: Broad disease spectrum and sex-specific phenotypes.

Authors:  Natalia N Singh; Shaine Hoffman; Prabhakara P Reddi; Ravindra N Singh
Journal:  Biochim Biophys Acta Mol Basis Dis       Date:  2021-01-05       Impact factor: 5.187

4.  Investigation on the Effects of Modifying Genes on the Spinal Muscular Atrophy Phenotype.

Authors:  Drenushe Zhuri; Hakan Gurkan; Damla Eker; Yasemin Karal; Sinem Yalcintepe; Engin Atli; Selma Demir; Emine Ikbal Atli
Journal:  Glob Med Genet       Date:  2022-09-05

Review 5.  In Search of a Cure: The Development of Therapeutics to Alter the Progression of Spinal Muscular Atrophy.

Authors:  Kristine S Ojala; Emily J Reedich; Christine J DiDonato; Stephen D Meriney
Journal:  Brain Sci       Date:  2021-02-05
  5 in total

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