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An Adult Zebrafish Model of Fibrodysplasia Ossificans Progressiva

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Bone Morphogenetic Proteins

Part of the book series: Methods in Molecular Biology ((MIMB,volume 1891))

Abstract

Fibrodysplasia ossificans progressiva (FOP) is a rare human skeletal disease caused by constitutively activating mutations in the gene ACVR1, which encodes a type I BMP/TGFβ family member receptor. FOP is characterized by progressive heterotopic ossification (HO) of fibrous tissues, including skeletal muscle, tendons, and ligaments, as well as malformation of the big toes, vertebral fusions, and osteochondromas. Surgical interventions in patients often result in enhanced HO, which can exacerbate rather than improve diagnostic outcomes. As a result of these difficulties, a variety of animal models are needed to study human FOP. Here we describe the methods for creating and characterizing zebrafish conditionally expressing Acvr1lQ204D, the first adult zebrafish model for FOP.

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Acknowledgments

This work was supported by NIH/NIDCR R01DE018043 (PCY), R21AR065761 (PCY), and NSF GRFP NS9344 (ML).

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Authors and Affiliations

  1. Program in Cell, Molecular, and Developmental Biology, Sackler School of Graduate Biomedical Sciences, Tufts University School of Medicine, Boston, MA, USA

    Melissa LaBonty & Pamela C. Yelick

  2. Department of Orthodontics, Division of Craniofacial and Molecular Genetics, Tufts University School of Dental Medicine, Boston, MA, USA

    Melissa LaBonty & Pamela C. Yelick

Authors
  1. Melissa LaBonty
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  2. Pamela C. Yelick
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Corresponding author

Correspondence to Pamela C. Yelick .

Editor information

Editors and Affiliations

  1. New Jersey Medical School, Rutgers Biomedical and Health Sciences, Newark, NJ, USA

    Melissa B. Rogers

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LaBonty, M., Yelick, P.C. (2019). An Adult Zebrafish Model of Fibrodysplasia Ossificans Progressiva. In: Rogers, M. (eds) Bone Morphogenetic Proteins. Methods in Molecular Biology, vol 1891. Humana Press, New York, NY. https://doi.org/10.1007/978-1-4939-8904-1_11

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  • DOI: https://doi.org/10.1007/978-1-4939-8904-1_11

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  • Publisher Name: Humana Press, New York, NY

  • Print ISBN: 978-1-4939-8903-4

  • Online ISBN: 978-1-4939-8904-1

  • eBook Packages: Springer Protocols

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