What Matters to Patients With Cleft Lip and/or Palate: An International Qualitative Study Informing the Development of the CLEFT-Q

Cleft Palate Craniofac J. 2018 Mar;55(3):442-450. doi: 10.1177/1055665617732854. Epub 2017 Dec 14.


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.

Keywords: cleft lip and/or palate; interpretive description; patient-reported outcomes; qualitative.

Publication types

  • Multicenter Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adaptation, Psychological
  • Adolescent
  • Child
  • Cleft Lip / psychology*
  • Cleft Lip / surgery*
  • Cleft Palate / psychology*
  • Cleft Palate / surgery*
  • Female
  • Humans
  • Interviews as Topic
  • Male
  • Patient Reported Outcome Measures*
  • Qualitative Research
  • Quality of Life
  • Young Adult