Concepts Important to Patients With Facial Differences: A Qualitative Study Informing a New Module of the FACE-Q for Children and Young Adults.

OBJECTIVE: The concepts important to children and young adults who undergo treatments for facial differences are not well-defined. Measurement of treatment outcomes from the patient's perspective is necessary to ensure goals of treatment are met. We aimed to identify concepts important to children and young adults with facial differences through a qualitative study. DESIGN: An interpretive description qualitative approach was followed. Semistructured interviews were conducted, transcribed verbatim, and coded using a line-by-line approach. Qualitative analysis led to the development of a conceptual framework of outcomes important to patients. SETTING: Interviews were conducted in Canada and the United Kingdom at home, by telephone, or in the hospital. PARTICIPANTS: Participants (N = 72) were recruited between May and June 2014 from craniofacial clinics at the Hospital for Sick Children (Toronto) and Great Ormond Street Hospital (London). Participants included anyone with a visible and/or functional facial difference aged 8 to 29 years and fluent in English, excluding patients with a cleft. The sample included 38 females and 34 males, with a mean age of 13.9 years, and included 28 facial conditions (11 facial paralysis, 18 ear anomalies, 26 skeletal conditions, and 17 soft tissue conditions). RESULTS: Analysis led to identification of important concepts within 4 overarching domains: facial appearance, facial function, adverse effects of treatment, and health-related quality of life (psychological, social, and school function). CONCLUSIONS: Our study provides an understanding of concepts important to children and young adults with facial differences.

Impact of Completing CLEFT-Q Scales That Ask About Appearance on Children and Young Adults: An International Study.

OBJECTIVE: To describe the impact of completing the CLEFT-Q appearance scales on patients with cleft lip and/or palate and to identify demographic and clinical characteristics and CLEFT-Q scores associated with reporting a negative impact. DESIGN: International cross-sectional survey. SETTING: Recruitment took place between October 2014 and November 2016 at 30 craniofacial clinics located in 12 countries. PATIENTS: Aged 8 to 29 years with cleft lip and/or palate. MAIN OUTCOME MEASURE(S): Participants were asked 4 questions to evaluate the impact of completing the field test version of a patient-reported outcome measure (the CLEFT-Q) that included 154 items, of which 79 (51%) asked about appearance (of the face, nose, nostrils, teeth, lips, jaws, and cleft lip scar). RESULTS: The sample included 2056 participants. Most participants liked answering the CLEFT-Q (88%) and the appearance questions (82%). After completing the appearance scales, most participants (77%) did not feel upset or unhappy about how they look, and they felt the same (67%) or better (23%) about their appearance after completing the questionnaire. Demographic and clinical variables associated with feeling unhappy/upset or worse about how they look included country of residence, female gender, more severe cleft, anticipating future cleft-specific surgeries, and reporting lower (ie, worse) scores on CLEFT-Q appearance and health-related quality-of-life scales. CONCLUSION: Most participants liked completing the CLEFT-Q, but a small minority reported a negative impact. When used in clinical practice, CLEFT-Q scale scores should be examined as soon as possible after completion in order that the clinical team might identify patients who might require additional support.

Computerised adaptive testing accurately predicts CLEFT-Q scores by selecting fewer, more patient-focused questions.

BACKGROUND: The International Consortium for Health Outcome Measurement (ICHOM) has recently agreed upon a core outcome set for the comprehensive appraisal of cleft care, which puts a greater emphasis on patient-reported outcome measures (PROMs) and, in particular, the CLEFT-Q. The CLEFT-Q comprises 12 scales with a total of 110 items, aimed to be answered by children as young as 8 years old. OBJECTIVE: In this study, we aimed to use computerised adaptive testing (CAT) to reduce the number of items needed to predict results for each CLEFT-Q scale. METHOD: We used an open-source CAT simulation package to run item responses over each of the full-length scales and its CAT counterpart at varying degrees of precision, estimated by standard error (SE). The mean number of items needed to achieve a given SE was recorded for each scale's CAT, and the correlations between results from the full-length scales and those predicted by the CAT versions were calculated. RESULTS: Using CATs for each of the 12 CLEFT-Q scales, we reduced the number of questions that participants needed to answer, that is, from 110 to a mean of 43.1 (range 34-60, SE < 0.55) while maintaining a 97% correlation between scores obtained with CAT and full-length scales. CONCLUSIONS: CAT is likely to play a fundamental role in the uptake of PROMs into clinical practice given the high degree of accuracy achievable with substantially fewer items.

Translation and cultural adaptation of the CLEFT-Q for use in Colombia, Chile, and Spain.

BACKGROUND: Cleft lip and/or palate (CL/P) is a common congenital craniofacial anomaly that may negatively affect an individual's appearance, health-related quality of life, or speech. In Spain, Colombia, and Chile the overall prevalence of CL/P ranges from 0.53 to 1.59 cases per 1000 live births. Currently, there is no patient-reported outcome (PRO) instrument that is specific for patients with CL/P. The CLEFT-Q is a new PRO instrument developed to measure outcomes of treatment in patients 8 to 29 years of age with CL/P. The aim of this study was to translate and culturally adapt the CLEFT-Q for use in Colombia, Chile, and Spain. METHODS: The CLEFT-Q was translated from English to 3 Spanish language varieties (Colombian, Chilean, and Spanish (Spain)) and Catalan. Translation and cultural adaptation guidelines set forth by the International Society for Pharmacoeconomics and Outcomes Research were followed. RESULTS: The field- test version of the CLEFT-Q consisted of 13 scales (total 154 items) measuring appearance, health-related quality of life, and facial function. Forward translations revealed 10 (7%) items that were difficult to translate into Chilean, and back translations identified 34 (22%) and 21 (13%) items whose meaning differed from the English version in at least 1 of the 3 Spanish varieties and Catalan respectively. Twenty-one participants took part in cognitive debriefing interviews. Participants were recruited from plastic surgery centres in Bogotá, Colombia (n = 4), Santiago, Chile (n = 7), and Barcelona, Spain (n = 10). Most participants were males (n = 14, 67%) and were diagnosed with CL/P (n = 17, 81%). Participants reported difficulty understanding 1 item in the Colombian, 1 item in the Spanish (Spain), and 11 items from the Catalan version. Comparison of the 3 Spanish varieties revealed 61 (40%) of the 154 items whose wording differed across the 3 Spanish versions. CONCLUSION: Translation and cultural adaptation processes provided evidence of transferability of the CLEFT-Q scales into 3 Spanish varieties and Catalan, as semantic, idiomatic, experiential, and conceptual equivalence of the items, instructions, and response options were achieved.