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Cited by 62Developmental Biology and Gene RegulationOsteoarthritis Treatment and MechanismsCongenital limb and hand anomalies

The zinc finger transcription factors Osr1 and Osr2 control synovial joint formation

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Gao, Yang, Lan, Yu, Liu, Han, Jiang, Rulang·2011·Developmental Biology
DOI 10.1016/j.ydbio.2011.01.018PMC PMC3057278
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Abstract

Synovial joints enable smooth articulations between different skeletal elements and are essential for the motility of vertebrates. Despite decades of extensive studies of the molecular and cellular mechanisms of limb and skeletal development, the molecular mechanisms governing synovial joint formation are still poorly understood. In particular, whereas several signaling pathways have been shown to play critical roles in joint maintenance, the mechanism controlling joint initiation is unknown. Here we report that Osr1 and Osr2, the mammalian homologs of the odd-skipped family of zinc finger transcription factors that are required for leg joint formation in Drosophila, are both strongly expressed in the developing synovial joint cells in mice. Whereas Osr1(-/-) mutant mice died at midgestation and Osr2(-/-) mutant mice had only subtle defects in synovial joint development, tissue-specific inactivation of Osr1 in the developing limb mesenchyme in Osr2(-/-) mutant mice caused fusion of multiple joints. We found that Osr1 and Osr2 function is required for maintenance of expression of signaling molecules critical for joint formation, including Gdf5, Wnt4 and Wnt9b. In addition, joint cells in the double mutants failed to upregulate expression of the articular cartilage marker gene Prg4. These data indicate that Osr1 and Osr2 function redundantly to control synovial joint formation.

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  • The conserved enhancer mm703 is involved in the regulation of the Osr2 gene expressed in the synovial joint connecting the metapterygium and radials.

    “This conserved enhancer is involved in the regulation of the Osr2 gene expressed in the synovial joint connecting the metapterygium and radials (Gao et al., 2011).”

    From Ancestral developmental potentials in early bony fish contributed to vertebrate water-to-land transition (2021) · cites this paper as Gao et al., 2011

    The Results section reports Osr2 expression in developing mouse synovial joints, supporting that part of the claim. Our copy does not identify the enhancer mm703, show that it regulates Osr2, or describe a joint between the metapterygium and radials. (LLM, conf=0.85)

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References 56 · Cited by 62

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Authors
Gao, Y., Lan, Y., Liu, H., & Jiang, R.
Affiliations
University of Rochester Medicine (US)
Corresponding author
Jiang, Rulang
ORCID
Venue
Developmental Biology
Publisher
Elsevier BV
Published
2011-04
Volume
352
Issue
1
Pages
83-91
ISSN
0012-1606 (ISSN-L)
DOI
10.1016/j.ydbio.2011.01.018
PMC
PMC3057278
OpenAlex
W1981068023
Cited by
62
References
56
Citations by year
2026: 4 · 2025: 4 · 2024: 4 · 2023: 4 · 2022: 5 · 2021: 3 · 2020: 6 · 2019: 2 · 2018: 6 · 2017: 2 · 2016: 2 · 2015: 7 · 2014: 4 · 2013: 3 · 2012: 3
FWCI
1.69
Type
journal article
Language
en
Open access
Licence
Locations
  • Developmental Biology (publishedVersion)
  • PubMed (publishedVersion)
  • Europe PMC (PubMed Central) (acceptedVersion)
  • PubMed Central (acceptedVersion)
Funders
NIH/NIDCR (R01DE013681, R01DE018401)
Topics
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Developmental Biology and Gene Regulation
  • Health Sciences › Medicine › Rheumatology › Osteoarthritis Treatment and Mechanisms
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Developmental Biology › Congenital limb and hand anomalies
Keywords
OSR1, Osr2, zinc finger transcription factor, limb mesenchyme, arthrodesis, GDF5, Wnt4, Wnt9b, articular cartilage, proteoglycan 4
MeSH terms
Proteoglycan 4, Animals, Cell Differentiation, Mice, Mutant Strains, Mutation, Proteoglycans, Transcription Factors, Signal Transduction, Zinc Fingers, Joint Capsule, Homeodomain Proteins, Gene Expression Regulation, Developmental, Integrases, Organogenesis, Wnt Proteins, beta Catenin, Mice

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