odd-skipped related 2 is required for fin chondrogenesis in zebrafish

Authors

  • Pui-Ying Lam,

    1. Massachusetts General Hospital, Department of Medicine, Nephrology Division, and Harvard Medical School Department of Genetics, Charlestown, Massachusetts
    Current affiliation:
    1. Program in Cellular and Molecular Biology, University of Wisconsin-Madison, Madison, WI
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    • These authors contributed equally to this work.

  • Caramai N. Kamei,

    1. Massachusetts General Hospital, Department of Medicine, Nephrology Division, and Harvard Medical School Department of Genetics, Charlestown, Massachusetts
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    • These authors contributed equally to this work.

  • Steve Mangos,

    1. Massachusetts General Hospital, Department of Medicine, Nephrology Division, and Harvard Medical School Department of Genetics, Charlestown, Massachusetts
    Current affiliation:
    1. Rush University Medical Center, Chicago, IL
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  • Sudha Mudumana,

    1. Massachusetts General Hospital, Department of Medicine, Nephrology Division, and Harvard Medical School Department of Genetics, Charlestown, Massachusetts
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  • Yan Liu,

    1. Massachusetts General Hospital, Department of Medicine, Nephrology Division, and Harvard Medical School Department of Genetics, Charlestown, Massachusetts
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  • Iain A. Drummond

    Corresponding author
    1. Massachusetts General Hospital, Department of Medicine, Nephrology Division, and Harvard Medical School Department of Genetics, Charlestown, Massachusetts
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ABSTRACT

Background: odd-skipped related 2 (osr2) encodes a vertebrate ortholog of the Drosophila odd-skipped zinc-finger transcription factor. Osr2 in mouse is required for proper palate, eyelid, and bone development. Zebrafish knock-down experiments have also suggested a role for osr2, along with its paralog osr1, in early pectoral fin specification and pronephric development. Results: We show here that osr2 has a specific function later in development, independent of osr1, in the regulation of sox9a expression and promoting fin chondrogenesis. mRNA in situ hybridization demonstrated osr2 expression in the developing floorplate and later during organogenesis in the pronephros and gut epithelium. In the pectoral fin buds, osr2 was specifically expressed in fin mesenchyme. osr2 knock down in zebrafish embryos disrupted both three and five zinc finger alternatively spliced osr2 isoforms and eliminated wild-type osr2 mRNA. osr2 morphants exhibited normal pectoral fin bud specification but exhibited defective fin chondrogenesis, with loss of differentiated chondrocytes. Defects in chondrogenesis were paralleled by loss of sox9a as well as subsequent col2a1 expression, linking osr2 function to essential regulators of chondrogenesis. Conclusions: The zebrafish odd-skipped related 2 gene regulates sox9a and col2a1 expression in chondrocyte development and is specifically required for zebrafish fin morphogenesis. Developmental Dynamics 242:1284–1292, 2013. © 2013 Wiley Periodicals, Inc.

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