Karen W Y Wong Riff1,2, Elena Tsangaris2, Timothy E E Goodacre3, Christopher R Forrest1, Jessica Lawson4, Andrea L Pusic5, Anne F Klassen6. 1. 1 Division of Plastic and Reconstructive Surgery, Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada. 2. 2 Department of Clinical Epidemiology and Biostatistics, McMaster University, Hamilton, Ontario, Canada. 3. 3 Spires Cleft Center, Oxford Radcliffe Children's Hospital, Oxford, United Kingdom. 4. 4 Ferkauf Graduate School of Psychology, Yeshiva University, Bronx, NY, USA. 5. 5 Plastic and Reconstructive Surgery, Memorial Sloan-Kettering Cancer Center, New York, NY, USA. 6. 6 Department of Pediatrics, McMaster University, Hamilton, Canada.
Abstract
OBJECTIVE: The goal of treatment for individuals with cleft lip and/or palate (CL/P) is to improve physical, psychological, and social health. Outcomes of treatment are rarely measured from the patient's perspective. The aim of the study was to develop a conceptual framework for a patient-reported outcome (PRO) instrument for individuals with clefts (CLEFT-Q) by developing an in-depth understanding of issues that individuals consider to be important. DESIGN: The qualitative methodology of interpretive description was used. Setting, Participants, and Intervention: We performed 136 individual in-depth interviews with participants with clefts of any age, presenting for cleft care, across 6 countries. Parents were involved if the child was more comfortable. Interviews were audio-recorded, transcribed verbatim, and coded using constant comparison. The data were used to develop a refined conceptual framework. RESULTS: Participants described concepts of interest in 3 top-level domains, each of which included subdomains: appearance (face, nose, nostrils, teeth, lips, jaw, cleft lip scar), health-related quality of life (psychological, social, school, speech-related distress), and facial function (speech, eating/drinking). Participants were able to describe changes over time with regard to the 3 domains. CONCLUSIONS: A conceptual framework of concepts of interest to individuals with CL/P formed the basis of the scales in the CLEFT-Q. Each subdomain represents an independently functioning scale. Understanding what matters to patients is essential in guiding PRO measurement.
OBJECTIVE: The goal of treatment for individuals with cleft lip and/or palate (CL/P) is to improve physical, psychological, and social health. Outcomes of treatment are rarely measured from the patient's perspective. The aim of the study was to develop a conceptual framework for a patient-reported outcome (PRO) instrument for individuals with clefts (CLEFT-Q) by developing an in-depth understanding of issues that individuals consider to be important. DESIGN: The qualitative methodology of interpretive description was used. Setting, Participants, and Intervention: We performed 136 individual in-depth interviews with participants with clefts of any age, presenting for cleft care, across 6 countries. Parents were involved if the child was more comfortable. Interviews were audio-recorded, transcribed verbatim, and coded using constant comparison. The data were used to develop a refined conceptual framework. RESULTS:Participants described concepts of interest in 3 top-level domains, each of which included subdomains: appearance (face, nose, nostrils, teeth, lips, jaw, cleft lip scar), health-related quality of life (psychological, social, school, speech-related distress), and facial function (speech, eating/drinking). Participants were able to describe changes over time with regard to the 3 domains. CONCLUSIONS: A conceptual framework of concepts of interest to individuals with CL/P formed the basis of the scales in the CLEFT-Q. Each subdomain represents an independently functioning scale. Understanding what matters to patients is essential in guiding PRO measurement.
Authors: Anne F Klassen; Karen Wy Wong Riff; Natasha M Longmire; Asteria Albert; Gregory C Allen; Mustafa Asim Aydin; Stephen B Baker; Stefan J Cano; Andrew J Chan; Douglas J Courtemanche; Marieke M Dreise; Jesse A Goldstein; Timothy E E Goodacre; Karen E Harman; Montserrat Munill; Aisling O Mahony; Mirta Palomares Aguilera; Petra Peterson; Andrea L Pusic; Rona Slator; Mia Stiernman; Elena Tsangaris; Sunil S Tholpady; Federico Vargas; Christopher R Forrest Journal: CMAJ Date: 2018-04-16 Impact factor: 8.262
Authors: Kristin D Pfeifauf; Alison K Snyder-Warwick; Sibyl Scheve; Cheryl L Grellner; Gary B Skolnick; Andrew Wilkey; Jordan Foy; Sybill D Naidoo; Kamlesh B Patel Journal: Cleft Palate Craniofac J Date: 2020-01-17
Authors: Elena Tsangaris; Karen W Y Wong Riff; Federico Vargas; Mirta Palomares Aguilera; Macarena Miranda Alarcón; Asteria Albert Cazalla; Lehana Thabane; Achilleas Thoma; Anne F Klassen Journal: Health Qual Life Outcomes Date: 2017-11-28 Impact factor: 3.186
Authors: Anne F Klassen; Natasha M Longmire; Neil W Bulstrode; David M Fisher; Leila Kasrai; Justine O'Hara; Vivek Panchapakesan; Andrea L Pusic; Ken Stewart; Elena Tsangaris; Natalia Ziolkowski; Karen W Y Wong Riff Journal: Plast Reconstr Surg Glob Open Date: 2018-08-07
Authors: Anne F Klassen; Natalia Ziolkowski; Lily R Mundy; H Catherine Miller; Alison McIlvride; Allison DiLaura; Joel Fish; Andrea L Pusic Journal: Plast Reconstr Surg Glob Open Date: 2018-04-24