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Injury of Adult Zebrafish Expressing Acvr1l Q204D Does Not Result in Heterotopic Ossification.
- Source :
-
Zebrafish [Zebrafish] 2018 Dec; Vol. 15 (6), pp. 536-545. Date of Electronic Publication: 2018 Sep 05. - Publication Year :
- 2018
-
Abstract
- Fibrodysplasia Ossificans Progressiva (FOP) is a rare, autosomal dominant genetic disorder in humans characterized by the gradual ossification of fibrous tissues, including skeletal muscle, tendons, and ligaments. In humans, mutations in the Type I BMP/TGFβ family member receptor gene, ACVR1, are associated with FOP. Zebrafish acvr1l, previously known as alk8, is the functional ortholog of human ACVR1. We previously created and characterized the first adult zebrafish model for FOP by generating animals harboring heat shock-inducible mCherry-tagged constitutively active Acvr1l (Q204D). Since injury is a known trigger for heterotopic ossification (HO) development in human FOP patients, in this study, we investigated several injury models in Acvr1l <superscript>Q204D</superscript> -expressing zebrafish and the subsequent formation of HO. We performed studies of Activin A injection, cardiotoxin (CTX) injection, and caudal fin clip injury. We found that none of these methods resulted in HO formation at the site of injury. However, some of the cardiotoxin-injected and caudal fin-clipped animals did exhibit HO at distant sites, including the body cavity and along the spine. We describe these results in the context of new and exciting reports on FOP, and discuss future studies to better understand the etiology and progression of this disease.
- Subjects :
- Activins administration & dosage
Activins genetics
Animal Fins injuries
Animal Fins metabolism
Animals
Animals, Genetically Modified genetics
Cardiotoxins administration & dosage
Humans
Zebrafish genetics
Zebrafish Proteins genetics
Activin Receptors, Type I metabolism
Animal Fins pathology
Animals, Genetically Modified physiology
Mutation
Ossification, Heterotopic physiopathology
Zebrafish physiology
Zebrafish Proteins metabolism
Subjects
Details
- Language :
- English
- ISSN :
- 1557-8542
- Volume :
- 15
- Issue :
- 6
- Database :
- MEDLINE
- Journal :
- Zebrafish
- Publication Type :
- Academic Journal
- Accession number :
- 30183553
- Full Text :
- https://doi.org/10.1089/zeb.2018.1611