Literature DB >> 26744358

Towards a European Registry and Biorepository for Patients with Spinal and Bulbar Muscular Atrophy.

Davide Pareyson1, Pietro Fratta2, Pierre-François Pradat3,4, Gianni Sorarù5, Josef Finsterer6, John Vissing7, Manu E Jokela8, Bjarne Udd9, Albert C Ludolph10, Anna Sagnelli11, Patrick Weydt12.   

Abstract

Pathomechanisms of spinal and bulbar muscular atrophy (SBMA) have been extensively investigated and are partially understood, but no effective treatment is currently available for this disabling disorder. Its rarity, the slow disease progression, and lack of sensitive-to-change outcome measures render design and conduction of clinical trials a challenging task. Therefore, it is fundamental to strengthen the network of clinical centers interested in SBMA for clinical trial readiness. We propose to create and maintain an International SBMA Registry where as many well-characterized patients as possible can be included, with the following aims: facilitate planning of clinical trials and recruitment of patients, define natural history of the disease, characterize epidemiology, develop standards of care, and inform the community of patients about research progresses and ongoing trials. We also aim at developing harmonized and coordinated biorepositories. The experience obtained during the last years in the field of other neuromuscular disorders and of Huntington disease offers valuable precedents.

Entities:  

Keywords:  Biorepository; Clinical trials; Kennedy disease; Registry; Spinal and bulbar muscular atrophy

Mesh:

Year:  2016        PMID: 26744358     DOI: 10.1007/s12031-015-0704-5

Source DB:  PubMed          Journal:  J Mol Neurosci        ISSN: 0895-8696            Impact factor:   3.444


  24 in total

1.  168th ENMC International Workshop: outcome measures and clinical trials in Charcot-Marie-Tooth disease (CMT).

Authors:  M M Reilly; M E Shy; F Muntoni; D Pareyson
Journal:  Neuromuscul Disord       Date:  2010-09-17       Impact factor: 4.296

2.  Observing Huntington's disease: the European Huntington's Disease Network's REGISTRY.

Authors:  Michael Orth; O J Handley; C Schwenke; S Dunnett; E J Wild; S J Tabrizi; G B Landwehrmeyer
Journal:  J Neurol Neurosurg Psychiatry       Date:  2010-11-19       Impact factor: 10.154

3.  210th ENMC International Workshop: Research and clinical management of patients with spinal and bulbar muscular atrophy, 27-29 March, 2015, Naarden, The Netherlands.

Authors:  Maria Pennuto; Linda Greensmith; Pierre-François Pradat; Gianni Sorarù
Journal:  Neuromuscul Disord       Date:  2015-06-19       Impact factor: 4.296

4.  If you build a rare disease registry, will they enroll and will they use it? Methods and data from the National Registry of Myotonic Dystrophy (DM) and Facioscapulohumeral Muscular Dystrophy (FSHD).

Authors:  James E Hilbert; John T Kissel; Elizabeth A Luebbe; William B Martens; Michael P McDermott; Donald B Sanders; Rabi Tawil; Charles A Thornton; Richard T Moxley
Journal:  Contemp Clin Trials       Date:  2011-11-26       Impact factor: 2.226

5.  Reliability and validity of the CMT neuropathy score as a measure of disability.

Authors:  M E Shy; J Blake; K Krajewski; D R Fuerst; M Laura; A F Hahn; J Li; R A Lewis; M Reilly
Journal:  Neurology       Date:  2005-04-12       Impact factor: 9.910

Review 6.  Clinical Trials in Spinal and Bulbar Muscular Atrophy-Past, Present, and Future.

Authors:  Patrick Weydt; Anna Sagnelli; Angela Rosenbohm; Pietro Fratta; Pierre-François Pradat; Albert C Ludolph; Davide Pareyson
Journal:  J Mol Neurosci       Date:  2015-11-14       Impact factor: 3.444

7.  Characterization of the DMD/BMD patient population in Czech Republic and Slovakia using an innovative registry approach.

Authors:  Petr Brabec; Petr Vondrácek; Daniel Klimes; Sarah Baumeister; Hanns Lochmüller; Tomás Pavlík; Jakub Gregor
Journal:  Neuromuscul Disord       Date:  2009-03-09       Impact factor: 4.296

8.  Riluzole in Huntington's disease: a 3-year, randomized controlled study.

Authors:  G Bernhard Landwehrmeyer; Bruno Dubois; Justo Garcia de Yébenes; Berry Kremer; Wilhelm Gaus; Peter H Kraus; Horst Przuntek; Michel Dib; Adam Doble; Wilhelm Fischer; Albert C Ludolph
Journal:  Ann Neurol       Date:  2007-09       Impact factor: 10.422

9.  157th ENMC International Workshop: patient registries for rare, inherited muscular disorders 25-27 January 2008 Naarden, The Netherlands.

Authors:  Anna Sárközy; Kate Bushby; Christophe Béroud; Hanns Lochmüller
Journal:  Neuromuscul Disord       Date:  2008-10-22       Impact factor: 4.296

10.  Characteristics of Japanese Duchenne and Becker muscular dystrophy patients in a novel Japanese national registry of muscular dystrophy (Remudy).

Authors:  Harumasa Nakamura; En Kimura; Madoka Mori-Yoshimura; Hirofumi Komaki; Yu Matsuda; Kanako Goto; Yukiko K Hayashi; Ichizo Nishino; Shin'ichi Takeda; Mitsuru Kawai
Journal:  Orphanet J Rare Dis       Date:  2013-04-19       Impact factor: 4.123

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  7 in total

1.  Introduction to the Special Issue on Spinal and Bulbar Muscular Atrophy.

Authors:  Maria Pennuto; Illana Gozes
Journal:  J Mol Neurosci       Date:  2016-02-13       Impact factor: 3.444

Review 2.  National information system for rare diseases with an approach to data architecture: A systematic review.

Authors:  Simin Derayeh; Alireza Kazemi; Reza Rabiei; Azamossadat Hosseini; Hamid Moghaddasi
Journal:  Intractable Rare Dis Res       Date:  2018-08

Review 3.  Kennedy's disease (spinal and bulbar muscular atrophy): a clinically oriented review of a rare disease.

Authors:  Marianthi Breza; Georgios Koutsis
Journal:  J Neurol       Date:  2018-07-13       Impact factor: 4.849

4.  Validation of the Italian version of the SBMA Functional Rating Scale as outcome measure.

Authors:  Giorgia Querin; Elisa DaRe; Ilaria Martinelli; Luca Bello; Cinzia Bertolin; Davide Pareyson; Caterina Mariotti; Elena Pegoraro; Gianni Sorarù
Journal:  Neurol Sci       Date:  2016-07-21       Impact factor: 3.307

5.  The French national protocol for Kennedy's disease (SBMA): consensus diagnostic and management recommendations.

Authors:  Pierre-François Pradat; Emilien Bernard; Philippe Corcia; Philippe Couratier; Christel Jublanc; Giorgia Querin; Capucine Morélot Panzini; François Salachas; Christophe Vial; Karim Wahbi; Peter Bede; Claude Desnuelle
Journal:  Orphanet J Rare Dis       Date:  2020-04-10       Impact factor: 4.123

6.  The Italian neuromuscular registry: a coordinated platform where patient organizations and clinicians collaborate for data collection and multiple usage.

Authors:  Anna Ambrosini; Daniela Calabrese; Francesco Maria Avato; Felice Catania; Guido Cavaletti; Maria Carmela Pera; Antonio Toscano; Giuseppe Vita; Lucia Monaco; Davide Pareyson
Journal:  Orphanet J Rare Dis       Date:  2018-10-04       Impact factor: 4.123

Review 7.  Biomarkers of Spinal and Bulbar Muscle Atrophy (SBMA): A Comprehensive Review.

Authors:  Giorgia Querin; Peter Bede; Veronique Marchand-Pauvert; Pierre-Francois Pradat
Journal:  Front Neurol       Date:  2018-10-10       Impact factor: 4.003

  7 in total

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