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马凡综合征的骨骼肌改变:一项系统综述

Skeletal muscle alterations in Marfan syndrome: a systematic review.

作者信息

Santos Audrei R, Gutierrez Rita M S, Koike Tatiana E, Conte Talita C, Real Caroline C, Dumont Nicolas A, Miyabara Elen H

机构信息

Department of Anatomy, Institute of Biomedical Sciences, University of São Paulo, São Paulo, Brazil.

CHU Sainte-Justine Research Center, Montreal, Canada.

出版信息

J Muscle Res Cell Motil. 2025 Aug 20. doi: 10.1007/s10974-025-09706-x.

Abstract

Marfan syndrome is an autosomal dominant multisystemic connective tissue disorder caused by mutations in the FBN1 gene. Although clinical changes in the cardiovascular, ocular, and skeletal systems have been described in detail in Marfan syndrome patients, investigations about skeletal muscle alterations are still incipient. This systematic review describes cellular, molecular, and functional changes in skeletal muscles of patients and mice with Marfan syndrome. Study selection (from EMBASE, MEDLINE, and Web of Science databases), data extraction, and quality appraisal were performed by two independent reviewers. A total of 2634 articles were identified; 26 were included in the analysis based on the selection criteria. The risk of bias was evaluated using the Critical Appraisal Skills Programme and Joanna Briggs Institute Critical Appraisal tool for human studies and the Systematic Review Centre for Laboratory Animal Experimentation RoB tool for animal studies. The findings indicate that skeletal muscle alterations in Marfan syndrome are characterized by fibrosis, reduced muscle mass and myofiber size, compromised muscle regeneration, and impaired muscle function. Future studies are warranted to investigate the mechanisms involved in the development of this muscle phenotype to help develop effective strategies to improve skeletal muscle function and the quality of life of individuals with Marfan syndrome.

摘要

马凡综合征是一种由FBN1基因突变引起的常染色体显性多系统结缔组织疾病。尽管马凡综合征患者心血管、眼部和骨骼系统的临床变化已得到详细描述,但关于骨骼肌改变的研究仍处于起步阶段。本系统综述描述了马凡综合征患者和小鼠骨骼肌的细胞、分子和功能变化。由两名独立评审员进行研究选择(来自EMBASE、MEDLINE和Web of Science数据库)、数据提取和质量评估。共识别出2634篇文章;根据选择标准,26篇文章被纳入分析。使用批判性评估技能计划和乔安娜·布里格斯研究所人类研究批判性评估工具以及实验室动物实验系统综述中心动物研究偏倚风险工具评估偏倚风险。研究结果表明,马凡综合征患者的骨骼肌改变表现为纤维化、肌肉质量和肌纤维大小减小、肌肉再生受损以及肌肉功能受损。有必要开展进一步研究,以探究这种肌肉表型发展过程中的相关机制,从而有助于制定有效策略,改善马凡综合征患者的骨骼肌功能和生活质量。

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