Content Validity of Patient-Reported Outcome Instruments used with Pediatric Patients with Facial Differences

Wickert, Natasha; Riff, Karen W. Y. Wong; Mansour, Mark; Forrest, Christopher R.; Goodacre, Tim; Pusic, Andrea L.; Klassen, Anne F.

doi:10.1597/16-148

Cited by 17 publications

(18 citation statements)

References 46 publications

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“…We previously identified the need for PRO instruments developed specifically for CL/P (Klassen et al, 2012). We also showed that existing PRO instruments used with children with facial differences lack content validity (Wickert et al, 2018). Content validity refers to whether content of a PRO instrument is relevant, comprehensive, and comprehensible (Terwee et al, 2018).…”

Section: Introductionmentioning

confidence: 81%

“…Tapia et al (2016) also reviewed PRO instruments used in craniofacial research and concluded that there was a need for PRO instruments for pediatric patients with diverse facial differences. An important methodological weakness of existing PRO instruments for patients with facial differences is that they were created without input from patients (Tapia et al, 2016;Wickert et al, 2018). Further, the content of the few PRO instruments designed for patients with facial differences tends to use negative phrasing or content (Wickert et al, 2018).…”

Section: Introductionmentioning

confidence: 99%

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

Kamran

Longmire

Rae

et al. 2020

The Cleft Palate Craniofacial Journal

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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.

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Section: Introductionmentioning

confidence: 81%

Section: Introductionmentioning

confidence: 99%

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

Kamran

Longmire

Rae

et al. 2020

The Cleft Palate Craniofacial Journal

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“… 18 Elsewhere, we report findings from our systematic review of pediatric PRO instruments developed for conditions associated with a facial difference. 8 …”

Section: Methodsmentioning

confidence: 99%

“…In plastic surgery, measurement of the patient’s perspective has been hampered by the lack of validated PRO instruments. Systematic reviews of pediatric PRO instruments for conditions associated with a facial difference identified only 1 ear-specific PRO measure, 1 , 8 , 9 that is, the Congenital Aural Atresia Questionnaire, which measures hearing and psychosocial concerns. 10 Given the lack of ear-specific PRO instruments, researchers have used generic measures.…”

Section: Introductionmentioning

confidence: 99%

Development of a New Patient-reported Outcome Measure for Ear Conditions: The EAR-Q

Klassen

Longmire

Bulstrode

et al. 2018

Plastic and Reconstructive Surgery - Global Open

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Background:Patient-reported outcome measures are widely used to improve health services and patient outcomes. The aim of our study was to describe the development of 2 ear-specific scales designed to measure outcomes important to children and young adults with ear conditions, such as microtia and prominent ears.Methods:We used an interpretive description qualitative approach. Semi-structured qualitative and cognitive interviews were performed with participants with any type of ear condition recruited from plastic surgery clinics in Canada, Australia, United States, and United Kingdom. Participants were interviewed to elicit new concepts. Interviews were audio-recorded, transcribed, and coded using the constant comparison approach. Experts in ear reconstruction were invited to provide input via an online Research Electronic Data Capture survey.Results:Participants included 25 patients aged 8–21 years with prominent ears (n = 9), microtia (n = 14), or another condition that affected ear appearance (n = 2). Analysis of participant qualitative data, followed by cognitive interviews and expert input, led to the development and refinement of an 18-item ear appearance scale (eg, size, shape, look up close, look in photographs) and a 12-item adverse effects scale (eg, itchy, painful, numb).Conclusions:The EAR-Q in currently being field-tested internationally. Once finalized, we anticipate the EAR-Q will be used in clinical practice and research to understand the patient’s perspective of outcomes following ear surgery.

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“…Beyond the scope of CL/P, few scales exist that measure appraisal of appearance from the patient perspective 10. Congenital anomalies, trauma and other benign and malignant conditions can cause facial or other differences that are stigmatising and may lead to social isolation.…”

Section: Introductionmentioning

confidence: 99%

International multiphase mixed methods study protocol to develop a cross-cultural patient-reported outcome instrument for children and young adults with cleft lip and/or palate (CLEFT-Q)

Riff

Tsangaris

Goodacre³

et al. 2017

BMJ Open

100

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IntroductionPatient-reported outcome (PRO) instruments should be developed according to rigorous guidelines in order to provide clinically meaningful, scientifically sound measurement. Understanding the methodology behind instrument development informs the selection of the most appropriate tool. This mixed methods protocol describes the development of an internationally applicable PRO instrument, the CLEFT-Q, for evaluating outcomes of treatment for cleft lip and/or palate (CL/P).Methods and analysisThe study includes three main phases that occur iteratively and interactively. In phase I, we determine what concepts are important to patients regarding their outcome. A conceptual framework for the CLEFT-Q is formed through a systematic review and an extensive international qualitative study. The systematic review ascertains what concepts have previously been measured in patients with CL/P. The qualitative study employs interpretive description and involves in-depth interviews with patients in high-income and lower-middle income countries. Preliminary items are generated from the qualitative data. Preliminary scales are then created for each theme in the framework. Cognitive debriefing interviews and expert clinician input are used to refine the scales in an iterative process. In phase II, the preliminary scales are administered to a large international group of patients with CL/P. The modern psychometric method of Rasch Measurement Theory analysis is employed to define the measurement characteristics. The preliminary scales are shortened based on these results. In phase III, further tests assess reliability, validity and responsiveness of the instrument.Ethics and disseminationThe study is approved by Research Ethics Boards for each participating site. Findings from this study will be published in open access peer-reviewed journals and presented at national and international conferences. Integrated knowledge translation is employed to engage stakeholders from the outset of the study. Successful execution of the CLEFT-Q will result in an internationally applicable PRO instrument for children and young adults with CL/P.

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Content Validity of Patient-Reported Outcome Instruments used with Pediatric Patients with Facial Differences

Abstract: 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.

Cited by 17 publications

References 46 publications

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

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

Development of a New Patient-reported Outcome Measure for Ear Conditions: The EAR-Q

International multiphase mixed methods study protocol to develop a cross-cultural patient-reported outcome instrument for children and young adults with cleft lip and/or palate (CLEFT-Q)

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