OBJECTIVES: We present an overview of the literature on caregiver stress in children with craniosynostosis and report common trends in the literature. INTRODUCTION: Craniosynostosis occurs approximately 1 in 2500 births. As this is a diagnosis most common in infants and often requires surgical treatment, this is a significant and stressful ordeal for caregivers. Caregiver stress impacts various outcomes for the child, and little is understood and known about caregiver stress in the pediatric craniosynostosis population. METHODS: A literature search for all articles pertaining to craniosynostosis and parental/caregiver stress was conducted using PubMed, Embase, PsychINFO, and CINAHL databases. RESULTS: Seven articles on caregiver stress in craniofacial abnormalities patients and three articles on caregiver stress in pediatric craniosynostosis patients specifically were identified. Three articles on parental satisfaction after craniosynostosis repair were also identified and included in the review. Few published studies exist in the literature on caregiver stress in children with craniosynostosis and no clear trends were identified. It is evident that caregiver stress significantly affects the psychosocial outcomes of children with craniosynostosis. However, there are an equal number of studies reporting significant differences in caregiver stress in children with craniosynostosis as those reporting no significant differences. CONCLUSIONS: There is evidence that caregiver stress affects psychosocial outcomes of children with craniosynostosis, but no clear trends of either increased or decreased levels of stress were identified in caregivers of children with craniosynostosis. Additional research is needed to identify risk factors related to caregiver stress.