Literature DB >> 34183144

Spinal Muscular Atrophy Treatments, Newborn Screening, and the Creation of a Neurogenetics Urgency.

Russell J Butterfield1.   

Abstract

Spinal muscular atrophy (SMA) is a progressive neuromuscular disorder characterized by loss of motor neurons leading to muscle weakness and atrophy. The United States' Food and Drug Administration's (FDA) approval of nusinersen, onasemnogene abeparvovec, and risdiplam for SMA has challenged existing treatment paradigms with multiple treatment options, a new natural history of the disease, and an emerging understanding of the importance of early and pre-symptomatic treatment. The profound impact of early, pre-symptomatic treatment has led to the creation of a neurogenetics urgency for newly identified patients with SMA, a novel problem for neurologists more accustomed to a more methodical approach to diagnosis and care. Implementation of newborn screening programs has helped facilitate early diagnosis and treatment, but challenges remain in overcoming administrative and procedural hurdles that can lead to treatment delays. Herein I discuss 2 cases that highlight the importance of early treatment, as well as gaps in our understanding of the progression of SMA in pre-symptomatic infants.
Copyright © 2021 Elsevier Inc. All rights reserved.

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Year:  2021        PMID: 34183144      PMCID: PMC8243405          DOI: 10.1016/j.spen.2021.100899

Source DB:  PubMed          Journal:  Semin Pediatr Neurol        ISSN: 1071-9091            Impact factor:   3.042


  37 in total

1.  Time is muscle: A recommendation for early treatment for preterm infants with spinal muscular atrophy.

Authors:  Bo Hoon Lee; Megan A Waldrop; Anne M Connolly; Emma Ciafaloni
Journal:  Muscle Nerve       Date:  2021-05-07       Impact factor: 3.217

2.  Diagnosis and management of spinal muscular atrophy: Part 2: Pulmonary and acute care; medications, supplements and immunizations; other organ systems; and ethics.

Authors:  Richard S Finkel; Eugenio Mercuri; Oscar H Meyer; Anita K Simonds; Mary K Schroth; Robert J Graham; Janbernd Kirschner; Susan T Iannaccone; Thomas O Crawford; Simon Woods; Francesco Muntoni; Brunhilde Wirth; Jacqueline Montes; Marion Main; Elena S Mazzone; Michael Vitale; Brian Snyder; Susana Quijano-Roy; Enrico Bertini; Rebecca Hurst Davis; Ying Qian; Thomas Sejersen
Journal:  Neuromuscul Disord       Date:  2017-11-23       Impact factor: 4.296

Review 3.  Diagnosis and management of spinal muscular atrophy: Part 1: Recommendations for diagnosis, rehabilitation, orthopedic and nutritional care.

Authors:  Eugenio Mercuri; Richard S Finkel; Francesco Muntoni; Brunhilde Wirth; Jacqueline Montes; Marion Main; Elena S Mazzone; Michael Vitale; Brian Snyder; Susana Quijano-Roy; Enrico Bertini; Rebecca Hurst Davis; Oscar H Meyer; Anita K Simonds; Mary K Schroth; Robert J Graham; Janbernd Kirschner; Susan T Iannaccone; Thomas O Crawford; Simon Woods; Ying Qian; Thomas Sejersen
Journal:  Neuromuscul Disord       Date:  2017-11-23       Impact factor: 4.296

4.  Risdiplam in Type 1 Spinal Muscular Atrophy.

Authors:  Giovanni Baranello; Basil T Darras; John W Day; Nicolas Deconinck; Andrea Klein; Riccardo Masson; Eugenio Mercuri; Kristy Rose; Muna El-Khairi; Marianne Gerber; Ksenija Gorni; Omar Khwaja; Heidemarie Kletzl; Renata S Scalco; Timothy Seabrook; Paulo Fontoura; Laurent Servais
Journal:  N Engl J Med       Date:  2021-02-24       Impact factor: 91.245

5.  Observational study of spinal muscular atrophy type I and implications for clinical trials.

Authors:  Richard S Finkel; Michael P McDermott; Petra Kaufmann; Basil T Darras; Wendy K Chung; Douglas M Sproule; Peter B Kang; A Reghan Foley; Michelle L Yang; William B Martens; Maryam Oskoui; Allan M Glanzman; Jean Flickinger; Jacqueline Montes; Sally Dunaway; Jessica O'Hagen; Janet Quigley; Susan Riley; Maryjane Benton; Patricia A Ryan; Megan Montgomery; Jonathan Marra; Clifton Gooch; Darryl C De Vivo
Journal:  Neurology       Date:  2014-07-30       Impact factor: 9.910

6.  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

7.  Prospective Cohort Study of Nusinersen Treatment in Adults with Spinal Muscular Atrophy.

Authors:  Crystal Jing Jing Yeo; Sarah D Simeone; Elise L Townsend; Ren Zhe Zhang; Kathryn J Swoboda
Journal:  J Neuromuscul Dis       Date:  2020

8.  Health outcomes in spinal muscular atrophy type 1 following AVXS-101 gene replacement therapy.

Authors:  Samiah Al-Zaidy; A Simon Pickard; Kavitha Kotha; Lindsay N Alfano; Linda Lowes; Grace Paul; Kathleen Church; Kelly Lehman; Douglas M Sproule; Omar Dabbous; Benit Maru; Katherine Berry; W David Arnold; John T Kissel; Jerry R Mendell; Richard Shell
Journal:  Pediatr Pulmonol       Date:  2018-12-12

Review 9.  Spinal Muscular Atrophy: Mutations, Testing, and Clinical Relevance.

Authors:  Melissa C Keinath; Devin E Prior; Thomas W Prior
Journal:  Appl Clin Genet       Date:  2021-01-25

Review 10.  Yeo and Darras: Extraneuronal Phenotypes of Spinal Muscular Atrophy.

Authors:  Crystal Jing Jing Yeo; Basil T Darras
Journal:  Ann Neurol       Date:  2020-10-29       Impact factor: 10.422

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