Literature DB >> 22516079

Childhood spinal muscular atrophy: controversies and challenges.

Eugenio Mercuri1, Enrico Bertini, Susan T Iannaccone.   

Abstract

Spinal muscular atrophy is an autosomal recessive disorder characterised by degeneration of motor neurons in the spinal cord and is caused by mutations of the survival of motor neuron 1 gene SMN1. The severity of spinal muscular atrophy is highly variable and no cure is available at present. Consensus has been reached on several aspects of care, the availability of which can have a substantial effect on prognosis, but controversies remain. The development of standards of care for children with the disorder and the identification of promising treatment strategies have changed the natural history of spinal muscular atrophy, and the prospects are good for further improvements in function, quality of life, and survival. A long-term benefit for patients will be the development of effective interventions (such as antisense oligonucleotides), some of which are in clinical trials. The need to be prepared for clinical trials has been the impetus for a remarkable and unprecedented cooperation between clinicians, scientists, industry, government, and volunteer organisations on an international scale.
Copyright © 2012 Elsevier Ltd. All rights reserved.

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Year:  2012        PMID: 22516079     DOI: 10.1016/S1474-4422(12)70061-3

Source DB:  PubMed          Journal:  Lancet Neurol        ISSN: 1474-4422            Impact factor:   44.182


  77 in total

Review 1.  Genetic approaches to the treatment of inherited neuromuscular diseases.

Authors:  Bhavya Ravi; Anthony Antonellis; Charlotte J Sumner; Andrew P Lieberman
Journal:  Hum Mol Genet       Date:  2019-10-01       Impact factor: 6.150

Review 2.  The promise of futility trials in neurological diseases.

Authors:  Marcus W Koch; Lawrence Korngut; David G Patry; Yahya Agha-Khani; Christopher White; Justyna R Sarna; Michael Yeung; V Wee Yong; Daniel Y C Heng; Gary Cutter; Luanne Metz
Journal:  Nat Rev Neurol       Date:  2015-03-17       Impact factor: 42.937

3.  Cost Effectiveness of Nusinersen in the Treatment of Patients with Infantile-Onset and Later-Onset Spinal Muscular Atrophy in Sweden.

Authors:  Santiago Zuluaga-Sanchez; Megan Teynor; Christopher Knight; Robin Thompson; Thomas Lundqvist; Mats Ekelund; Annabelle Forsmark; Adrian D Vickers; Andrew Lloyd
Journal:  Pharmacoeconomics       Date:  2019-06       Impact factor: 4.981

4.  Drug treatment for spinal muscular atrophy types II and III.

Authors:  Renske I Wadman; W Ludo van der Pol; Wendy Mj Bosboom; Fay-Lynn Asselman; Leonard H van den Berg; Susan T Iannaccone; Alexander Fje Vrancken
Journal:  Cochrane Database Syst Rev       Date:  2020-01-06

5.  Natural history of infantile-onset spinal muscular atrophy.

Authors:  Stephen J Kolb; Christopher S Coffey; Jon W Yankey; Kristin Krosschell; W David Arnold; Seward B Rutkove; Kathryn J Swoboda; Sandra P Reyna; Ai Sakonju; Basil T Darras; Richard Shell; Nancy Kuntz; Diana Castro; Julie Parsons; Anne M Connolly; Claudia A Chiriboga; Craig McDonald; W Bryan Burnette; Klaus Werner; Mathula Thangarajh; Perry B Shieh; Erika Finanger; Merit E Cudkowicz; Michelle M McGovern; D Elizabeth McNeil; Richard Finkel; Susan T Iannaccone; Edward Kaye; Allison Kingsley; Samantha R Renusch; Vicki L McGovern; Xueqian Wang; Phillip G Zaworski; Thomas W Prior; Arthur H M Burghes; Amy Bartlett; John T Kissel
Journal:  Ann Neurol       Date:  2017-12-08       Impact factor: 10.422

6.  Electrocardiographic Tremor as an Important Diagnostic Aid in Spinal Muscular Atrophy.

Authors:  Hansashree Padmanabha; Razia Adam Kadwa; Jitendra Kumar Sahu; Pratibha D Singhi
Journal:  Indian J Pediatr       Date:  2016-07-27       Impact factor: 1.967

7.  Quantitative muscle ultrasound measures rapid declines over time in children with SMA type 1.

Authors:  Kay W Ng; Anne M Connolly; Craig M Zaidman
Journal:  J Neurol Sci       Date:  2015-08-28       Impact factor: 3.181

8.  Motor neuron disease: A prospective natural history study of type 1 spinal muscular atrophy.

Authors:  Enrico Bertini; Eugenio Mercuri
Journal:  Nat Rev Neurol       Date:  2018-01-19       Impact factor: 42.937

9.  SMA valiant trial: a prospective, double-blind, placebo-controlled trial of valproic acid in ambulatory adults with spinal muscular atrophy.

Authors:  John T Kissel; Bakri Elsheikh; Wendy M King; Miriam Freimer; Charles B Scott; Stephen J Kolb; Sandra P Reyna; Thomas O Crawford; Louise R Simard; Kristin J Krosschell; Gyula Acsadi; Mary K Schroth; Guy D'Anjou; Bernard LaSalle; Thomas W Prior; Susan Sorenson; Jo Anne Maczulski; Kathryn J Swoboda
Journal:  Muscle Nerve       Date:  2014-02       Impact factor: 3.217

10.  Intrathecal administration of Nusinersen in type 1 SMA: successful psychological program in a single Italian center.

Authors:  Stefania La Foresta; Cristina Faraone; Maria Sframeli; Gian Luca Vita; Massimo Russo; Claudia Profazio; Immacolata Rulli; Eloisa Gitto; Antonio Versaci; Sonia Messina; Giuseppe Vita
Journal:  Neurol Sci       Date:  2018-07-25       Impact factor: 3.307

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