Background: The advance in imaging techniques is useful for 3D models and printing leading to a real revolution in many surgical specialties, in particular, neurosurgery. Methods: We report on a clinical study on the use of 3D printed models to perform cranioplasty in patients with craniosynostosis. The participants were recruited from various medical institutions and were divided into two groups: Group A (n= 5) received traditional surgical education (including cadaveric specimens) but without using 3D printed models, while Group B (n= 5) received training using 3D printed models. Results: Group B surgeons had the opportunity to plan different techniques and to simulate the cranioplasty. Group B surgeons reported that models provided a realistic and controlled environment for practicing surgical techniques, allowed for repetitive practice, and helped in visualizing the anatomy and pathology of craniosynostosis. Conclusion: 3D printed models can provide a realistic and controlled environment for neurosurgeons to develop their surgical skills in a safe and efficient manner. The ability to practice on 3D printed models before performing the actual surgery on patients may potentially improve the surgeons’ confidence and competence in performing complex craniosynostosis surgeries.