Background: Craniosynostosis, the premature closure of skull sutures, can cause a misshapen head and neurologic issues. Surgical correction may result in bone defects requiring additional surgery. Case Description: We present a 5-year-old female who underwent cranioplasty for a skull defect and bulging brain after craniosynostosis surgery. Cranioplasty corrected the defect's functional and cosmetic aspects. CT with 3D reconstruction confirmed findings and aided planning. The patient underwent cranioplasty using mesh over the defect and had an unremarkable recovery. Result: A 5-year-old had residual craniosynostosis bone defect and bulging brain. Cranioplasty used mesh over the defect, had unremarkable recovery. Early intervention and a multidisciplinary approach are key. More research is needed on this condition's prevalence, causes and management. Conclusion: This case emphasizes early intervention and a multidisciplinary approach for craniosynostosis and related defects. A team approach addressing the condition's physical and psychological impacts on the child and family is crucial for optimal outcomes. Cranioplasty is critical, and success depends on expertise. This case highlights the importance of timely, appropriate craniosynostosis management and related defect care. We hope this report contributes to literature and improves outcomes for patients undergoing cranioplasty after craniosynostosis surgery.