Genetic regulation of injury-induced heterotopic ossification in adult zebrafish.
Arun-Kumar Kaliya-PerumalCenk CelikThomas James CarneyMatthew P HarrisPhilip W InghamPublished in: Disease models & mechanisms (2024)
Heterotopic ossification is the inappropriate formation of bone in soft tissues of the body. It can manifest spontaneously in rare genetic conditions or as a response to injury, known as acquired heterotopic ossification. There are several experimental models for studying acquired heterotopic ossification from different sources of damage. However, their tenuous mechanistic relevance to the human condition, invasive and laborious nature and/or lack of amenability to chemical and genetic screens, limit their utility. To address these limitations, we developed a simple zebrafish injury model that manifests heterotopic ossification with high penetrance in response to clinically emulating injuries, as observed in human myositis ossificans traumatica. Using this model, we defined the transcriptional response to trauma, identifying differentially regulated genes. Mutant analyses revealed that an increase in the activity of the potassium channel Kcnk5b potentiates injury response, whereas loss of function of the interleukin 11 receptor paralogue (Il11ra) resulted in a drastically reduced ossification response. Based on these findings, we postulate that enhanced ionic signalling, specifically through Kcnk5b, regulates the intensity of the skeletogenic injury response, which, in part, requires immune response regulated by Il11ra.
Keyphrases
- genome wide
- endothelial cells
- immune response
- gene expression
- rheumatoid arthritis
- transcription factor
- dna methylation
- induced pluripotent stem cells
- oxidative stress
- high throughput
- disease activity
- high glucose
- high intensity
- dendritic cells
- bone mineral density
- soft tissue
- single cell
- toll like receptor
- systemic lupus erythematosus
- body composition
- postmenopausal women
- drug induced
- heat shock
- binding protein
- wild type
- solid state