COSMIN database

Content Validity of Patient-Reported Outcome Instruments used with Pediatric Patients with Facial Differences: A Systematic Review

Wickert, N. M., Wong Riff, K. W. Y., Mansour, M., Forrest, C. R., Goodacre, T. E. E., Pusic, A. L., and Klassen, A. F.
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.
Cleft Palate Craniofac J
Publication year:
Symptom status:
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
Children (0-18)
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
PRO / non-PRO:
Patient Reported Outcome
Type of measurement instrument:
1 - Questionnaires
Acne-QoL - Acne-Specific Quality of Life Instrument
Acne-QOLI - Acne Quality of Life Index
APSEA - Assessment of the Psychological and Social Effects of Acne
BOQ - Burn Outcomes Questionnaire (versions: BOQ 0-5 years; BOQ 5-18 years; BOQ10-18 years; BOQ parent form 0-5 years; BOQ 5-18 years parent form; BOQ 11-18 adolescent form)
CAAQ - Congenital Aural Atresia Questionnaire
CADI - Cardiff Acne Disability Index
CDLQI - Children's Dermatology Life Quality Index
CFEQ - Craniofacial Experiences Questionnaire
CHQ - Child Health Questionnaire
COHIP - Child Oral Health Impact Profile
CPQ11–14 - Child Perceptions Questionnaire 11–14
CPQ8-10 - Child Perceptions Questionnaire 8–10
KIDSCREEN-27 items - Health Related Quality of Life Questionnaire for Children and Young People and their Parents-27 items
PedsQL - Pediatric Quality of Life Inventory
TACQOL - Netherlands Organization for Applied Scientific Research Academic Medical Center-Child Quality of Life   
YQOL-FD - Youth Quality of Life-Facial Differences Module
YQOL - Youth Quality of Life Instrument