Work

Myomaker and Myomixer are required for craniofacial myoblast fusion in zebrafish

Year: 2025

Type: article

Abstract: BACKGROUND: Craniofacial and trunk skeletal muscles are derived from different progenitor populations during development. Trunk skeletal muscles contain mostly multinucleated myofibers that are formed through myoblast fusion. However, myoblast fusion in craniofacial muscles and its molecular regulation are not well understood. Recent studies revealed that genetic mutations in MYOMAKER and MYOMIXER fusogens in humans cause Carey-Fineman-Ziter Syndrome (CFZS), characterized by facial weakness and lower jaw deformity. RESULTS: Previous studies in zebrafish revealed that knockout of myomaker and myomixer resulted in deformed craniofacial formation. To establish the causal connection between loss of fusogen function and craniofacial deformities, we characterized myoblast fusion in zebrafish craniofacial muscles. Our results demonstrate that myomaker and myomixer are expressed in both slow and fast craniofacial muscles, and loss of these fusogens results in defects in craniofacial myoblast fusion. Interestingly, unlike trunk muscles of early embryos and larvae that show fast-fiber-specific fusogen expression and fusion while slow muscle fusion only occurs at 3 weeks post-fertilization, both slow and fast craniofacial muscles fuse as early as 3 days post-fertilization. CONCLUSIONS: Collectively, this study demonstrates that myomaker and myomixer are expressed in both slow and fast-twitch craniofacial muscles and are essential for myoblast fusion and the development of craniofacial muscles.

Source: Developmental Dynamics

DOI: 10.1002/dvdy.70031

Authors: Zhanxiong Zhang, Pengzheng Yong, Qiaomu Hu, Shaojun Du

Institutions: University of Maryland, Baltimore, Institute of Marine and Environmental Technology, Yellow Sea Fisheries Research Institute, Chinese Academy of Fishery Sciences

Language: English

Topic: Muscle Physiology and Disorders

Subfield: Molecular Biology

Field: Biochemistry, Genetics and Molecular Biology

Domain: Life Sciences

Open Access status: hybrid

Estimated APC paid: $4,430

Funders: China Scholarship Council, National Institute of Arthritis and Musculoskeletal and Skin Diseases