| First Author | Decker-Rockefeller B | Year | 2023 |
| Journal | Animal Model Exp Med | Volume | 6 |
| Issue | 4 | Pages | 362-368 |
| PubMed ID | 37682010 | Mgi Jnum | J:358635 |
| Mgi Id | MGI:7782691 | Doi | 10.1002/ame2.12343 |
| Citation | Decker-Rockefeller B, et al. (2023) Whole mount of adult ear skin as a model to study vascular malformations. Animal Model Exp Med 6(4):362-368 |
| abstractText | BACKGROUND: Genetic analysis in human patients has linked mutations in PIK3CA, the catalytic subunit of PI-3'Kinase, to sporadic incidences of vascular malformations. METHODS: We have developed a mouse model with inducible and endothelial-specific expression of PIK3CA(H1047R) , resulting in the development of vascular malformations. Systemic induction of this mutation in adult mice results in rapid lethality, limiting our ability to track and study these lesions; therefore, we developed a topical and local induction protocol using the active metabolite of tamoxifen, 4OH-T, on the ear skin of adults. RESULTS: This approach allows us to successfully model the human disease in a mature and established vascular bed and track the development of vascular malformations. To validate the utility of this model, we applied a topical rapamycin ointment, as rapamycin is therapeutically beneficial to patients in clinical trials. We found that the induced ear lesions showed significant attenuation after treatment, which was easily quantified. CONCLUSIONS: These data collectively provide evidence of a new model to study vascular malformations in adult tissues, which should be particularly useful in environments lacking specialized small-animal imaging facilities. |
