Skip to content

Scleraxis genes are required for normal musculoskeletal development and for rib growth and mineralization in zebrafish

Research output: Contribution to journalArticle

Original languageEnglish
Pages (from-to)9116-9130
Number of pages15
JournalFASEB Journal
Volume33
Issue number8
Early online date17 May 2019
DOIs
DateAccepted/In press - 15 Apr 2019
DateE-pub ahead of print - 17 May 2019
DatePublished (current) - 1 Aug 2019

Abstract

Tendons are an essential part of the musculoskeletal system, connecting muscle and skeletal elements to enable force generation. The transcription factor scleraxis marks vertebrate tendons from early specification. Scleraxis-null mice are viable and have a range of tendon and bone defects in the trunk and limbs but no described cranial phenotype. We report the expression of zebrafish scleraxis orthologs: scleraxis homolog (scx)-a and scxb in cranial and intramuscular tendons and in other skeletal elements. Single mutants for either scxa or scxb, generated by clustered regularly interspaced short palindromic repeats (CRISPR)/CRISPR-associated protein 9 (Cas9), are viable and fertile as adult fish. Although scxb mutants show no obvious phenotype, scxa mutant embryos have defects in cranial tendon maturation and muscle misalignment. Mutation of both scleraxis genes results in more severe defects in cranial tendon differentiation, muscle and cartilage dysmorphogenesis and paralysis, and lethality by 2-5 wk, which indicates an essential function of scleraxis for craniofacial development. At juvenile and adult stages, ribs in scxa mutants fail to mineralize and/or are small and heavily fractured. Scxa mutants also have smaller muscle volume, abnormal swim movement, and defects in bone growth and composition. Scleraxis function is therefore essential for normal craniofacial form and function and vital for fish development.-Kague, E., Hughes, S. M., Lawrence, E. A., Cross, S., Martin-Silverstone, E., Hammond, C. L., Hinits, Y. Scleraxis genes are required for normal musculoskeletal development and for rib growth and mineralization in zebrafish.

    Research areas

  • craniofacial, muscle, ribs, tendon, zebrafish

Download statistics

No data available

Documents

Documents

  • Full-text PDF (final published version)

    Rights statement: This is the final published version of the article (version of record). It first appeared online via Federation of American Society of Experimental Biology at https://www.fasebj.org/doi/full/10.1096/fj.201802654RR . Please refer to any applicable terms of use of the publisher.

    Final published version, 2.99 MB, PDF document

    Licence: CC BY

DOI

View research connections

Related faculties, schools or groups