ABSTRACT Craniosynostosis is a debilitating congenic condition characterized by premature cranial suture fusion, resulting in abnormal skull shape, blindness, and potential neurologic and/or developmental defects. The prevalence of craniosynostosis is at approximately 1 in 2,500 live births, which is one of the highest incidences of congenital malformations of the skeletal system. We have assembled a strong collaborative team combining Dr. Hatch’s expertise in the mechanisms underlying coronal craniosynostosis and Dr. Mishina’s expertise in suture development and midline craniosynostosis animal model along with experiences in 3D tissue engineering scaffolds and biomolecule delivery for craniofacial tissue regeneration. In the parental grant, we have proposed the following specific Aims: Aim 1, we will develop 3D scaffold-based microenvironment to maintain suture mesenchymal stem cells; in Aim 2, we will establish 3D culture system to regenerate and maintain bone- suture-bone tissue composite; in Aim 3, we will establish the efficacy of engineered scaffolds and controlled release systems for preventing craniosynostosis in vivo. By accomplishing these specific aims, we have developed a novel strategy to cure or diminish the severity and recurrence of craniosynostosis in humans, hence significantly improving patient quality of life. During 3 and half years of the funding period of the parent grant, we have made substantial progresses on the Aim 1 and Aim 2. We are currently putting more efforts towards Aim 3 experiments, which requires development of a survival animal surgery model. Addition of Ms. Jaylynn Jones will further accelerate our team efforts to propel our progress and enhance the diversity of our interdisciplinary team, while providing her meaningful career development opportunity.