ABSTRACT
INTRODUCTION: Craniofacial surgeons treat patients with diverse craniofacial conditions (CFCs). Yet, little is known about the health-related quality of life (HRQoL) impact of diverse CFCs. Currently, there are no suitable instruments that measure the HRQoL of patients with diverse CFCs from the perspective of children and parents. The objective of this study was to develop the items and support the content validity of a comprehensive patient and parent-reported outcomes measure. METHODS: An iterative process consisting of a systematic literature review, expert opinion and in-depth interviews with patients and parents of patients with diverse CFCs was used. The literature review and expert opinion were used to generate in-depth interview questions. We interviewed 127 subjects: 80 parents of patients ages 0 to 18 years or older and 47 patients ages 7 to 18 years or older. English and Spanish speakers were represented in our sample. The majority of subjects originated from the United States and Mexico (83%). Craniofacial conditions included were cleft lip/palate, craniosynostosis, craniofacial microsomia, microtia, and dermatological conditions. Semistructured interviews were conducted until content saturation was achieved. Line-by-line analysis of interview transcripts identified HRQoL themes. Themes were interpreted and organized into larger domains that represent the conceptual framework of CFC-associated HRQoL. Themes were operationalized into items that represent the HRQoL issues of patients for both parent and patient versions. RESULTS: Six final bilingual and bicultural scales based on the domains derived from the literature review, expert opinion, and in-depth interviews were developed: (1) "Social Impact," (2) "Psychological Function," (3) "Physical Function," (4) "Family Impact," (5) "Appearance," And (6) "Finding Meaning." Some cultural differences were identified: in contrast to children from Mexico and other developing nations, families from the United States did not report public harassment or extremely negative public reactions to patients' CFC. Religion and spirituality were common themes in interviews of Spanish-speaking subjects but less common in interviews of English-speaking subjects. CONCLUSIONS: Qualitative methods involving pediatric patients with diverse CFCs and their parents in the item development process support the content validity for this bilingual and bicultural HRQoL instrument. The items developed in this study will now undergo psychometric testing in national multisite studies for validation.
