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A longitudinal study of CMT1A using Rasch analysis based CMT neuropathy and examination scores by Vera Fridman; Stefan Sillau; Gyula Acsadi; Chelsea Bacon; Kimberly Dooley; Joshua Burns; John Day; Shawna Feely; Richard S. Finkel; Tiffany Grider; Laurie Gutmann; David N. Herrmann; Callyn A. Kirk; Sarrah A. Knause; Matilde Laurá; Richard A. Lewis; Jun Li; Thomas E. Lloyd; Isabella Moroni; Francesco Muntoni; Emanuela Pagliano; Chiara Pisciotta; Giuseppe Piscosquito; Sindhu Ramchandren; Mario Saporta; Reza Sadjadi; Rosemary R. Shy; Carly E. Siskind; Charlotte J. Sumner; David Walk; Janel Wilcox; Sabrina W. Yum; Stephan Züchner; Steven S. Scherer; Davide Pareyson; Mary M. Reilly; Michael E. Shy; the Inherited Neuropathies Consortium—Rare Diseases Clinical Research Network (INC-RDCRN) is a Medicine article available to read on EtoBox.
What is A longitudinal study of CMT1A using Rasch analysis based CMT neuropathy and examination scores about?
## Objective To evaluate the sensitivity of Rasch analysis-based, weighted Charcot-Marie-Tooth Neuropathy and Examination Scores (CMTNS-R and CMTES-R) to clinical progression in patients with Charcot-Marie-Tooth disease type 1A (CMT1A). ## Methods Patients with CMT1A from 18 sites of the Inherited Neuropathies Consortium were evaluated between 2009 and 2018. Weighted CMTNS and CMTES modified category responses were developed with Rasch analysis of the standard scores. Change from baseline for CMTNS-R and CMTES-R was estimated with longitudinal regression models. ## Results Baseline CMTNS-R and CMTES-R scores were available for 517 and 1,177 participants, respectively. Mean ± SD age of participants with available CMTES-R scores was 41 ± 18 (range 4–87) years, and 56% were female. Follow-up CMTES-R assessments at 1, 2, and 3 years were available for 377, 321, and 244 patients. A mixed regression model showed significant change in CMTES-R score at years 2 through 6 compared to baseline (mean change from baseline 0.59 points at 2 years, p = 0.0004, n = 321). Compared to the original CMTES, the CMTES-R revealed a 55% improvement in the standardized response mean (mean change/SD change)
Who reads A longitudinal study of CMT1A using Rasch analysis based CMT neuropathy and examination scores?
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- Author
- Vera Fridman; Stefan Sillau; Gyula Acsadi; Chelsea Bacon; Kimberly Dooley; Joshua Burns; John Day; Shawna Feely; Richard S. Finkel; Tiffany Grider; Laurie Gutmann; David N. Herrmann; Callyn A. Kirk; Sarrah A. Knause; Matilde Laurá; Richard A. Lewis; Jun Li; Thomas E. Lloyd; Isabella Moroni; Francesco Muntoni; Emanuela Pagliano; Chiara Pisciotta; Giuseppe Piscosquito; Sindhu Ramchandren; Mario Saporta; Reza Sadjadi; Rosemary R. Shy; Carly E. Siskind; Charlotte J. Sumner; David Walk; Janel Wilcox; Sabrina W. Yum; Stephan Züchner; Steven S. Scherer; Davide Pareyson; Mary M. Reilly; Michael E. Shy; the Inherited Neuropathies Consortium—Rare Diseases Clinical Research Network (INC-RDCRN)
- Publisher
- Lippincott Williams and Wilkins; Ovid Technologies (Wolters Kluwer) - American Academy of Neurology; Lippincott Williams & Wilkins Ltd.; Ovid Technologies (Wolters Kluwer Health) (ISSN 0028-3878)
- Published
- 2020
- Language
- EN
- Field
- Medicine (Health Sciences)