Modeling of Dysferlinopathy (LGMDR2) Progression Article Swipe

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C. Bolano-Díaz , Harmen Reyngoudt , Ian Wilson , M. James , Fiona Elizabeth Smith , Ericky Caldas de Almeida Araújo , Heather Gordish‐Dressman , H. Hilsden , Laura Rufibach , DJ Wallace , L. Ward , Roberto Stramare , Alessandro Rampado , Mark Smith , Jean‐Marc Boisserie , Julien Le Louër , Sheryl Foster , Anthony Peduto , Noriko Sato , Takeshi Tamaru , Anne Marie Sawyer , John Day , Kristi Jones , Maggie C. Walter , Tanya Stojkovic , Madoka Mori‐Yoshimura , Jerry R. Mendell , Elena Pegoraro , Volker Straub , Andrew M. Blamire , Pierre G. Carlier , Jordi Díaz‐Manera , Marni Jacobs , Adrienne Arrieta , Eui Whan Hwang , Elaine R. Lee , Brent Yetter , Sabine Krause , Olivia Schreiber‐Katz , Elizabeth Harris , Michela Guglieri , Teresinha Evangelista , A. St. J. Murphy , Michelle Eagle , Robert Muni‐Lofra , D. Moat , Jassi Sodhi , Helen Sutherland , Tim Hodgson , Chiara Calore , Claudio Semplicini , Luca Bello , Jean Yves , Oumar Diabaté , Jennifer Pérez , Carolina Tesi Rocha , Richard Gee , Nigel F. Clarke , Sarah A. Sandaradura , Roula Ghaoui , Kayla Cornett , Claire A. Miller , Shin'ich Takeda , Ai Ashida , Takayuki Tateishi , Hiroyuki Yajima , Takahiro Nakayama , Kazuhiko Segawa , Masamori Endo ·

YOU? · · 2025 · Open Access · · DOI: https://doi.org/10.1212/nxg.0000000000200283

In conclusion, this study provides a better understanding of skeletal muscle fat replacement progression in the lower limb muscles of patients with LGMDR2, highlighting the influence of age at symptom onset, sex, and baseline motor function, which should be considered in the design and analysis of clinical trials. Although complex models improved the overall data fit, they did not improve the accuracy in identifying changes at a patient level, underlying the need for further research and validation and the fact that other variables we have not measured are probably influencing progression.

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Medicine Multivariate analysis Internal medicine Multivariate statistics Muscular dystrophy Cardiology Clinical trial Physical medicine and rehabilitation Physical therapy Statistics Mathematics

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Type: article
Language: en
Landing Page: https://doi.org/10.1212/nxg.0000000000200283
PDF: https://www.neurology.org/doi/pdf/10.1212/NXG.0000000000200283
OA Status: gold
Cited By: 1
References: 32
Related Works: 10
OpenAlex ID: https://openalex.org/W4412723461

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DOI: https://doi.org/10.1212/nxg.0000000000200283

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Title: Modeling of Dysferlinopathy (LGMDR2) Progression

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Type: article

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Language: en

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Publication year: 2025

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Publication date: 2025-07-29

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Authors: C. Bolano-Díaz, Harmen Reyngoudt, Ian Wilson, M. James, Fiona Elizabeth Smith, Ericky Caldas de Almeida Araújo, Heather Gordish‐Dressman, H. Hilsden, Laura Rufibach, DJ Wallace, L. Ward, Roberto Stramare, Alessandro Rampado, Mark Smith, Jean‐Marc Boisserie, Julien Le Louër, Sheryl Foster, Anthony Peduto, Noriko Sato, Takeshi Tamaru, Anne Marie Sawyer, John Day, Kristi Jones, Maggie C. Walter, Tanya Stojkovic, Madoka Mori‐Yoshimura, Jerry R. Mendell, Elena Pegoraro, Volker Straub, Andrew M. Blamire, Pierre G. Carlier, Jordi Díaz‐Manera, Marni Jacobs, Adrienne Arrieta, Eui Whan Hwang, Elaine R. Lee, Brent Yetter, Sabine Krause, Olivia Schreiber‐Katz, Elizabeth Harris, Michela Guglieri, Teresinha Evangelista, A. St. J. Murphy, Michelle Eagle, Robert Muni‐Lofra, D. Moat, Jassi Sodhi, Helen Sutherland, Tim Hodgson, Chiara Calore, Claudio Semplicini, Luca Bello, Jean Yves, Oumar Diabaté, Jennifer Pérez, Carolina Tesi Rocha, Richard Gee, Nigel F. Clarke, Sarah A. Sandaradura, Roula Ghaoui, Kayla Cornett, Claire A. Miller, Shin'ich Takeda, Ai Ashida, Takayuki Tateishi, Hiroyuki Yajima, Takahiro Nakayama, Kazuhiko Segawa, Masamori Endo

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Landing page: https://doi.org/10.1212/nxg.0000000000200283

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Concepts: Medicine, Multivariate analysis, Internal medicine, Multivariate statistics, Muscular dystrophy, Cardiology, Clinical trial, Physical medicine and rehabilitation, Physical therapy, Statistics, Mathematics

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Cited by: 1

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