Content Validity of Patient-Reported Outcome Instruments used with Pediatric Patients with Facial Differences: A Systematic Review
Authors:
Wickert, N. M., Wong Riff, K. W. Y., Mansour, M., Forrest, C. R., Goodacre, T. E. E., Pusic, A. L., and Klassen, A. F.
Abstract:
Objective The aim of this systematic review was to identify patient-reported outcome (PRO) instruments used in research with children/youth with conditions associated with facial differences to identify the health concepts measured. Design MEDLINE, EMBASE, CINAHL, and PsycINFO were searched from 2004 to 2016 to identify PRO instruments used in acne vulgaris, birthmarks, burns, ear anomalies, facial asymmetries, and facial paralysis patients. We performed a content analysis whereby the items were coded to identify concepts and categorized as positive or negative content or phrasing. Results A total of 7,835 articles were screened; 6 generic and 11 condition-specific PRO instruments were used in 96 publications. Condition-specific instruments were for acne (four), oral health (two), dermatology (one), facial asymmetries (two), microtia (one), and burns (one). The PRO instruments provided 554 items (295 generic; 259 condition specific) that were sorted into 4 domains, 11 subdomains, and 91 health concepts. The most common domain was psychological (n = 224 items). Of the identified items, 76% had negative content or phrasing (e.g., "Because of the way my face looks I wish I had never been born"). Given the small number of items measuring facial appearance (n = 19) and function (n = 22), the PRO instruments reviewed lacked content validity for patients whose condition impacted facial function and/or appearance. Conclusions Treatments can change facial appearance and function. This review draws attention to a problem with content validity in existing PRO instruments. Our team is now developing a new PRO instrument called FACE-Q Kids to address this problem.
Cognitive/mental state Emotional state Physical state
Functional status:
Physical functioning Role functioning Social functioning
General health perceptions / HRQoL:
Health-related quality of life Self-rated health
Age:
Children (0-18)
Disease:
Congenital malformations, deformations and chromosomal abnormalities and related symptoms Diseases of and symptoms related to the nervous system Diseases of and symptoms related to the skin and subcutaneous tissue Injury, poisoning and certain other consequences of external causes
