What Matters to Patients With Nonsyndromic Craniosynostosis and Their Parents: A Qualitative Study Informing the Development of a Patient-Reported Outcome Measures Set.

Abstract

The primary objective of this study was to identify important concepts that hold significance for both patients with nonsyndromic craniosynostosis and their parents regarding their craniosynostosis care. The findings from this study will guide the development of patient-reported outcome measures (PROM) sets tailored to specific craniosynostosis subtypes and age groups, which will be implemented in our treatment protocol. A qualitative methodology was used, and data were obtained through focus groups. A purposive sample of patients and parents of patients with nonsyndromic craniosynostosis was collected at Sophia Children's Hospital Erasmus Medical Center. The focus groups were recorded, transcribed verbatim, and analyzed using thematic content analysis. Important domains in the preoperative and postoperative period were identified. In total, 34 participants were included in this study, of which 24 parents and 10 patients were divided over 12 focus groups. Based on our qualitative data, 4 top-level domains were considered to be of importance in patients with nonsyndromic craniosynostosis and their parents, including: emotional and social, cognitive, physical, and disease-specific functioning. Each top-level domain consisted of subdomains. Findings from this qualitative study reveal domains important to patients with nonsyndromic craniosynostosis and their parents with regard to their outcomes in craniosynostosis care. The comprehensive framework derived from this study serves as a guideline for developing a craniosynostosis-specific PROM set of relevance to our population.