Literature DB >> 33412266

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

Natalia N Singh1, Shaine Hoffman2, Prabhakara P Reddi3, Ravindra N Singh4.   

Abstract

Spinal muscular atrophy (SMA) is one of the major genetic disorders associated with infant mortality. More than 90% of cases of SMA result from deletions of or mutations in the Survival Motor Neuron 1 (SMN1) gene. SMN2, a nearly identical copy of SMN1, does not compensate for the loss of SMN1 due to predominant skipping of exon 7. The spectrum of SMA is broad, ranging from prenatal death to infant mortality to survival into adulthood. All tissues, including brain, spinal cord, bone, skeletal muscle, heart, lung, liver, pancreas, gastrointestinal tract, kidney, spleen, ovary and testis, are directly and/or indirectly affected in SMA. Accumulating evidence on impaired mitochondrial biogenesis and defects in X chromosome-linked modifying factors, coupled with the sexual dimorphic nature of many tissues, point to sex-specific vulnerabilities in SMA. Here we review the role of sex in the pathogenesis of SMA.
Copyright © 2020 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Intronic splicing silencer N1 (ISS-N1); Male infertility; Mitochondria; Spinal muscular atrophy (SMA); Survival motor neuron (SMN); X chromosome

Mesh:

Substances:

Year:  2021        PMID: 33412266      PMCID: PMC7867633          DOI: 10.1016/j.bbadis.2020.166063

Source DB:  PubMed          Journal:  Biochim Biophys Acta Mol Basis Dis        ISSN: 0925-4439            Impact factor:   5.187


  275 in total

Review 1.  Advances in therapeutic development for spinal muscular atrophy.

Authors:  Matthew D Howell; Natalia N Singh; Ravindra N Singh
Journal:  Future Med Chem       Date:  2014-06       Impact factor: 3.808

2.  Smooth muscle atrophy and colon pathology in SMN deficient mice.

Authors:  Yun Yang; George Vassilakos; David W Hammers; Zhaohui Yang; Elisabeth R Barton; Hugh Lee Sweeney
Journal:  Am J Transl Res       Date:  2019-03-15       Impact factor: 4.060

3.  Defective neuromuscular junction organization and postnatal myogenesis in mice with severe spinal muscular atrophy.

Authors:  Elisabet Dachs; Marta Hereu; Lídia Piedrafita; Anna Casanovas; Jordi Calderó; Josep E Esquerda
Journal:  J Neuropathol Exp Neurol       Date:  2011-06       Impact factor: 3.685

4.  Type I spinal muscular atrophy can mimic sensory-motor axonal neuropathy.

Authors:  Evdokia Anagnostou; Steven P Miller; Marie-Christine Guiot; Greoge Karpati; Louise Simard; Marie-Emmanuelle Dilenge; Michael I Shevell
Journal:  J Child Neurol       Date:  2005-02       Impact factor: 1.987

5.  A single administration of morpholino antisense oligomer rescues spinal muscular atrophy in mouse.

Authors:  Paul N Porensky; Chalermchai Mitrpant; Vicki L McGovern; Adam K Bevan; Kevin D Foust; Brain K Kaspar; Stephen D Wilton; Arthur H M Burghes
Journal:  Hum Mol Genet       Date:  2011-12-20       Impact factor: 6.150

Review 6.  Estrogen actions in the brain and the basis for differential action in men and women: a case for sex-specific medicines.

Authors:  Glenda E Gillies; Simon McArthur
Journal:  Pharmacol Rev       Date:  2010-04-14       Impact factor: 25.468

7.  Spectrum of neuropathophysiology in spinal muscular atrophy type I.

Authors:  Brian N Harding; Shingo Kariya; Umrao R Monani; Wendy K Chung; Maryjane Benton; Sabrina W Yum; Gihan Tennekoon; Richard S Finkel
Journal:  J Neuropathol Exp Neurol       Date:  2015-01       Impact factor: 3.685

8.  Comprehensive nutritional and metabolic assessment in patients with spinal muscular atrophy: Opportunity for an individualized approach.

Authors:  Enid E Martinez; Nicolle Quinn; Kayla Arouchon; Rocco Anzaldi; Stacey Tarrant; Nina S Ma; John Griffin; Basil T Darras; Robert J Graham; Nilesh M Mehta
Journal:  Neuromuscul Disord       Date:  2018-03-19       Impact factor: 4.296

9.  Vascular Defects and Spinal Cord Hypoxia in Spinal Muscular Atrophy.

Authors:  Eilidh Somers; Robert D Lees; Katie Hoban; James N Sleigh; Haiyan Zhou; Francesco Muntoni; Kevin Talbot; Thomas H Gillingwater; Simon H Parson
Journal:  Ann Neurol       Date:  2016-01-13       Impact factor: 10.422

10.  Immune dysregulation may contribute to disease pathogenesis in spinal muscular atrophy mice.

Authors:  Marc-Olivier Deguise; Yves De Repentigny; Emily McFall; Nicole Auclair; Subash Sad; Rashmi Kothary
Journal:  Hum Mol Genet       Date:  2017-02-15       Impact factor: 6.150

View more
  4 in total

Review 1.  Structural Context of a Critical Exon of Spinal Muscular Atrophy Gene.

Authors:  Natalia N Singh; Collin A O'Leary; Taylor Eich; Walter N Moss; Ravindra N Singh
Journal:  Front Mol Biosci       Date:  2022-07-01

2.  Commentary: Current Status of Gene Therapy for Spinal Muscular Atrophy.

Authors:  Wilfried Rossoll; Ravindra N Singh
Journal:  Front Cell Neurosci       Date:  2022-05-17       Impact factor: 6.147

3.  Internal Introns Promote Backsplicing to Generate Circular RNAs from Spinal Muscular Atrophy Gene.

Authors:  Diou Luo; Natalia Nikolaevna Singh; Ravindra Narayan Singh
Journal:  Genes (Basel)       Date:  2022-06-25       Impact factor: 4.141

4.  Cerebrospinal Fluid and Clinical Profiles in Adult Type 2-3 Spinal Muscular Atrophy Patients Treated with Nusinersen: An 18-Month Single-Centre Experience.

Authors:  Giammarco Milella; Alessandro Introna; Eustachio D'Errico; Angela Fraddosio; Gaspare Scaglione; Antonella Morea; Maria Ucci; Maddalena Ruggieri; Mariangela Mastrapasqua; Marisa Megna; Filomena Puntillo; Isabella Laura Simone
Journal:  Clin Drug Investig       Date:  2021-08-13       Impact factor: 2.859

  4 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.