Literature DB >> 27701745

Revised upper limb module for spinal muscular atrophy: Development of a new module.

Elena S Mazzone1, Anna Mayhew2, Jacqueline Montes3, Danielle Ramsey4, Lavinia Fanelli1, Sally Dunaway Young3, Rachel Salazar3, Roberto De Sanctis1, Amy Pasternak5, Allan Glanzman6, Giorgia Coratti1, Matthew Civitello7, Nicola Forcina1, Richard Gee8, Tina Duong8, Marika Pane1, Mariacristina Scoto4, Maria Carmela Pera1, Sonia Messina9, Gihan Tennekoon6, John W Day8, Basil T Darras5, Darryl C De Vivo3, Richard Finkel7, Francesco Muntoni4, Eugenio Mercuri1.   

Abstract

INTRODUCTION: There is a growing need for a robust clinical measure to assess upper limb motor function in spinal muscular atrophy (SMA), as the available scales lack sensitivity at the extremes of the clinical spectrum. We report the development of the Revised Upper Limb Module (RULM), an assessment specifically designed for upper limb function in SMA patients.
METHODS: An international panel with specific neuromuscular expertise performed a thorough review of scales currently available to assess upper limb function in SMA. This review facilitated a revision of the existing upper limb function scales to make a more robust clinical scale.
RESULTS: Multiple revisions of the scale included statistical analysis and captured clinically relevant changes to fulfill requirements by regulators and advocacy groups.
CONCLUSIONS: The resulting RULM scale shows good reliability and validity, making it a suitable tool to assess upper extremity function in the SMA population for multi-center clinical research. Muscle Nerve 55: 869-874, 2017.
© 2016 Wiley Periodicals, Inc.

Entities:  

Keywords:  Hammersmith functional motor scale; Rasch analysis; functional assessment; outcome measures; spinal muscular atrophy; upper limb function

Mesh:

Year:  2017        PMID: 27701745     DOI: 10.1002/mus.25430

Source DB:  PubMed          Journal:  Muscle Nerve        ISSN: 0148-639X            Impact factor:   3.217


  40 in total

1.  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

2.  Quality of life assessment in adult spinal muscular atrophy patients treated with nusinersen.

Authors:  Silvia Bonanno; Riccardo Zanin; Luca Bello; Irene Tramacere; Virginia Bozzoni; Luca Caumo; Manfredi Ferraro; Sara Bortolani; Gianni Sorarù; Mauro Silvestrini; Veria Vacchiano; Mara Turri; Raffaella Tanel; Rocco Liguori; Michela Coccia; Renato Emilio Mantegazza; Tiziana Mongini; Elena Pegoraro; Lorenzo Maggi
Journal:  J Neurol       Date:  2022-01-03       Impact factor: 4.849

3.  Nusinersen Treatment in Adults With Spinal Muscular Atrophy.

Authors:  Tina Duong; Connie Wolford; Michael P McDermott; Chelsea E Macpherson; Amy Pasternak; Allan M Glanzman; William B Martens; Elizabeth Kichula; Basil T Darras; Darryl C De Vivo; Zarazuela Zolkipli-Cunningham; Richard S Finkel; Michael Zeineh; Max Wintermark; Jacinda Sampson; Katharine A Hagerman; Sally Dunaway Young; John W Day
Journal:  Neurol Clin Pract       Date:  2021-06

4.  Biomarkers of therapeutic efficacy in adolescents and adults with 5q spinal muscular atrophy: a systematic review.

Authors:  Maria Gavriilaki; Vasileios Papaliagkas; Alexandra Stamperna; Maria Moschou; Konstantinos Notas; Sotirios Papagiannopoulos; Marianthi Arnaoutoglou; Vasilios K Kimiskidis
Journal:  Acta Neurol Belg       Date:  2022-07-21       Impact factor: 2.471

Review 5.  Spinal muscular atrophy.

Authors:  Eugenio Mercuri; Charlotte J Sumner; Francesco Muntoni; Basil T Darras; Richard S Finkel
Journal:  Nat Rev Dis Primers       Date:  2022-08-04       Impact factor: 65.038

Review 6.  Pharmacotherapy for Spinal Muscular Atrophy in Babies and Children: A Review of Approved and Experimental Therapies.

Authors:  Claudia A Chiriboga
Journal:  Paediatr Drugs       Date:  2022-08-27       Impact factor: 3.930

Review 7.  Disease Modifying Therapies for the Management of Children with Spinal Muscular Atrophy (5q SMA): An Update on the Emerging Evidence.

Authors:  Helgi Thor Hjartarson; Kristofer Nathorst-Böös; Thomas Sejersen
Journal:  Drug Des Devel Ther       Date:  2022-06-16       Impact factor: 4.319

8.  Is cerebrospinal fluid amyloid-β42 a promising biomarker of response to nusinersen in adult spinal muscular atrophy patients?

Authors:  Alessandro Introna; Giammarco Milella; Eustachio D'Errico; Angela Fraddosio; Gaspare Scaglione; Maria Ucci; Maddalena Ruggieri; Isabella Laura Simone
Journal:  Muscle Nerve       Date:  2021-03-13       Impact factor: 3.217

9.  Nusinersen Wearing-Off in Adult 5q-Spinal Muscular Atrophy Patients.

Authors:  Alma Osmanovic; Olivia Schreiber-Katz; Susanne Petri
Journal:  Brain Sci       Date:  2021-03-13

10.  Nusinersen in pediatric and adult patients with type III spinal muscular atrophy.

Authors:  Maria Carmela Pera; Giorgia Coratti; Francesca Bovis; Marika Pane; Amy Pasternak; Jacqueline Montes; Valeria A Sansone; Sally Dunaway Young; Tina Duong; Sonia Messina; Irene Mizzoni; Adele D'Amico; Matthew Civitello; Allan M Glanzman; Claudio Bruno; Francesca Salmin; Simone Morando; Roberto De Sanctis; Maria Sframeli; Laura Antonaci; Anna Lia Frongia; Annemarie Rohwer; Mariacristina Scoto; Darryl C De Vivo; Basil T Darras; John Day; William Martens; Katia A Patanella; Enrico Bertini; Francesco Muntoni; Richard Finkel; Eugenio Mercuri
Journal:  Ann Clin Transl Neurol       Date:  2021-06-24       Impact factor: 4.511

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