Iyer Aishwarya, Alizadeh Madeline, Mariano Jennifer Megan, Kontrogianni-Konstantopoulos Aikaterini, Raufman Jean-Pierre
Department of Biochemistry and Molecular Biology, University of Maryland School of Medicine, Baltimore, Maryland.
The Institute for Genome Sciences, University of Maryland School of Medicine, Baltimore, Maryland.
Cell Mol Gastroenterol Hepatol. 2025 Apr 22;19(8):101522. doi: 10.1016/j.jcmgh.2025.101522.
Among other contributions to gastrointestinal (GI) function, skeletal muscles regulate transit at both ends of the GI tract by providing propulsive forces for ingested nutrients and controlling the excretion of waste products. At the oropharynx, skeletal muscles provide necessary forces for effective mastication and the transfer of food boluses from the mouth into the proximal esophagus, where skeletal muscle-mediated peristalsis initiates propulsion of food boluses towards the stomach, a function supplanted by the upper esophagus smooth muscle. Consequently, the most prominent manifestation of proximal GI tract skeletal muscle dysfunction is transfer and oropharyngeal dysphagia that may result in repeated episodes of life-threatening choking and pulmonary aspiration. At the anal canal, the external anal sphincter controls the release of gas, liquids, and solids. Skeletal muscles within the pelvic floor play a synergistic role in regulating defection. Hence, distal GI tract skeletal muscle dysfunction may result in the leakage of flatus and fecal matter, whereas, in contrast, pelvic floor dysfunction may contribute to constipation. The balance between such defects may severely impact nutritional status and quality of life. Herein, we provide a comprehensive review of the genetics, molecular biology, and mechanisms underlying heritable disorders of skeletal muscle and how these may impact GI tract function and overall well-being. For organizational purposes, we separate discussions of congenital, mitochondrial, and myofibrillar myopathies and muscular dystrophies. For the sake of completeness, we also briefly consider acquired myopathies that affect GI tract function. As treatment options are currently limited, disorders of skeletal muscle function provide exciting therapeutic opportunities, including innovative approaches to target specific gene modifications.
骨骼肌对胃肠(GI)功能有诸多贡献,它通过为摄入的营养物质提供推进力以及控制废物排泄来调节胃肠道两端的运输。在口咽部,骨骼肌为有效咀嚼以及将食团从口腔转移至食管近端提供必要的力量,在食管近端,骨骼肌介导的蠕动启动食团向胃的推进,而这一功能在食管上段被平滑肌所取代。因此,近端胃肠道骨骼肌功能障碍最显著的表现是转移和口咽性吞咽困难,这可能导致危及生命的呛噎和肺误吸反复发作。在肛管,肛门外括约肌控制气体、液体和固体的排出。盆底内的骨骼肌在调节排便中起协同作用。因此,远端胃肠道骨骼肌功能障碍可能导致肠胃气胀和粪便泄漏,而相比之下,盆底功能障碍可能导致便秘。这些缺陷之间的平衡可能严重影响营养状况和生活质量。在此,我们全面综述了骨骼肌遗传性疾病的遗传学、分子生物学及潜在机制,以及这些疾病如何影响胃肠道功能和整体健康。为便于组织讨论,我们将先天性、线粒体和肌原纤维肌病以及肌肉营养不良分开论述。为全面起见,我们还简要考虑了影响胃肠道功能的获得性肌病。由于目前治疗选择有限,骨骼肌功能障碍提供了令人兴奋的治疗机会,包括针对特定基因修饰的创新方法。