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The value of serum creatinine as biomarker of disease progression in spinal and bulbar muscular atrophy (SBMA)

Serum creatinine has been indicated as a potential marker of motor function in SBMA and results form previous longitudinal studies pointed to its decline over time. This is a longitudinal retrospective study investigating creatinine changes over a 36-month-period in 73 patients with SBMA. Severity a...

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Autores principales: Blasi, Lorenzo, Sabbatini, Daniele, Fortuna, Andrea, Querin, Giorgia, Martinelli, Ilaria, Vianello, Sara, Bertolin, Cinzia, Pareyson, Davide, Pennuto, Maria, Pegoraro, Elena, Bello, Luca, Sorarù, Gianni
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group UK 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10570332/
https://www.ncbi.nlm.nih.gov/pubmed/37828349
http://dx.doi.org/10.1038/s41598-023-44419-6
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author Blasi, Lorenzo
Sabbatini, Daniele
Fortuna, Andrea
Querin, Giorgia
Martinelli, Ilaria
Vianello, Sara
Bertolin, Cinzia
Pareyson, Davide
Pennuto, Maria
Pegoraro, Elena
Bello, Luca
Sorarù, Gianni
author_facet Blasi, Lorenzo
Sabbatini, Daniele
Fortuna, Andrea
Querin, Giorgia
Martinelli, Ilaria
Vianello, Sara
Bertolin, Cinzia
Pareyson, Davide
Pennuto, Maria
Pegoraro, Elena
Bello, Luca
Sorarù, Gianni
author_sort Blasi, Lorenzo
collection PubMed
description Serum creatinine has been indicated as a potential marker of motor function in SBMA and results form previous longitudinal studies pointed to its decline over time. This is a longitudinal retrospective study investigating creatinine changes over a 36-month-period in 73 patients with SBMA. Severity and progression of the disease was assessed according to serum creatine kinase (CK) values, manual muscle testing (MMT), SBMA functional rating scale (SBMAFRS) score, 6-min-walk test (6MWT) value, and spirometry (forced vital capacity, fVC%) obtained at the baseline and at each of the annual follow-up visits. Baseline serum creatinine concentrations positively correlated with 6MWT, the MMT megascore score of both the upper (ULM) and lower (LLM) limbs and SBMAFRS. No correlation was found with CK or fVC% values. Similar correlation results were achieved at all the subsequent time points. Longitudinal assessments conducted by the generalized estimating equations (GEE) method returned significant changes for SBMAFRS (− 1.41 points per year, p < 0.001), ULM and LLM (− 0.69, p = 0.01; and − 1.07, p < 0.001, respectively), 6MWT (− 47 m, p < 0.001) but not for creatinine (− 0.82, p > 0.05). We also observed that creatinine levels at baseline did not correlate with changes in the other measures from baseline at each annual visit. Our data do not support a role for serum creatinine as sensitive biomarker of disease progression, and possibily prognosis, in SBMA.
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spelling pubmed-105703322023-10-14 The value of serum creatinine as biomarker of disease progression in spinal and bulbar muscular atrophy (SBMA) Blasi, Lorenzo Sabbatini, Daniele Fortuna, Andrea Querin, Giorgia Martinelli, Ilaria Vianello, Sara Bertolin, Cinzia Pareyson, Davide Pennuto, Maria Pegoraro, Elena Bello, Luca Sorarù, Gianni Sci Rep Article Serum creatinine has been indicated as a potential marker of motor function in SBMA and results form previous longitudinal studies pointed to its decline over time. This is a longitudinal retrospective study investigating creatinine changes over a 36-month-period in 73 patients with SBMA. Severity and progression of the disease was assessed according to serum creatine kinase (CK) values, manual muscle testing (MMT), SBMA functional rating scale (SBMAFRS) score, 6-min-walk test (6MWT) value, and spirometry (forced vital capacity, fVC%) obtained at the baseline and at each of the annual follow-up visits. Baseline serum creatinine concentrations positively correlated with 6MWT, the MMT megascore score of both the upper (ULM) and lower (LLM) limbs and SBMAFRS. No correlation was found with CK or fVC% values. Similar correlation results were achieved at all the subsequent time points. Longitudinal assessments conducted by the generalized estimating equations (GEE) method returned significant changes for SBMAFRS (− 1.41 points per year, p < 0.001), ULM and LLM (− 0.69, p = 0.01; and − 1.07, p < 0.001, respectively), 6MWT (− 47 m, p < 0.001) but not for creatinine (− 0.82, p > 0.05). We also observed that creatinine levels at baseline did not correlate with changes in the other measures from baseline at each annual visit. Our data do not support a role for serum creatinine as sensitive biomarker of disease progression, and possibily prognosis, in SBMA. Nature Publishing Group UK 2023-10-12 /pmc/articles/PMC10570332/ /pubmed/37828349 http://dx.doi.org/10.1038/s41598-023-44419-6 Text en © The Author(s) 2023 https://creativecommons.org/licenses/by/4.0/Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) .
spellingShingle Article
Blasi, Lorenzo
Sabbatini, Daniele
Fortuna, Andrea
Querin, Giorgia
Martinelli, Ilaria
Vianello, Sara
Bertolin, Cinzia
Pareyson, Davide
Pennuto, Maria
Pegoraro, Elena
Bello, Luca
Sorarù, Gianni
The value of serum creatinine as biomarker of disease progression in spinal and bulbar muscular atrophy (SBMA)
title The value of serum creatinine as biomarker of disease progression in spinal and bulbar muscular atrophy (SBMA)
title_full The value of serum creatinine as biomarker of disease progression in spinal and bulbar muscular atrophy (SBMA)
title_fullStr The value of serum creatinine as biomarker of disease progression in spinal and bulbar muscular atrophy (SBMA)
title_full_unstemmed The value of serum creatinine as biomarker of disease progression in spinal and bulbar muscular atrophy (SBMA)
title_short The value of serum creatinine as biomarker of disease progression in spinal and bulbar muscular atrophy (SBMA)
title_sort value of serum creatinine as biomarker of disease progression in spinal and bulbar muscular atrophy (sbma)
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10570332/
https://www.ncbi.nlm.nih.gov/pubmed/37828349
http://dx.doi.org/10.1038/s41598-023-44419-6
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