The Development, Deployment, and Evaluation of the CLEFT-Q Computerized Adaptive Test: A Multimethods Approach Contributing to Personalized, Person-Centered Health Assessments in Plastic Surgery.

BACKGROUND: Routine use of patient-reported outcome measures (PROMs) and computerized adaptive tests (CATs) may improve care in a range of surgical conditions. However, most available CATs are neither condition-specific nor coproduced with patients and lack clinically relevant score interpretation. Recently, a PROM called the CLEFT-Q has been developed for use in the treatment of cleft lip or palate (CL/P), but the assessment burden may be limiting its uptake into clinical practice. OBJECTIVE: We aimed to develop a CAT for the CLEFT-Q, which could facilitate the uptake of the CLEFT-Q PROM internationally. We aimed to conduct this work with a novel patient-centered approach and make source code available as an open-source framework for CAT development in other surgical conditions. METHODS: CATs were developed with the Rasch measurement theory, using full-length CLEFT-Q responses collected during the CLEFT-Q field test (this included 2434 patients across 12 countries). These algorithms were validated in Monte Carlo simulations involving full-length CLEFT-Q responses collected from 536 patients. In these simulations, the CAT algorithms approximated full-length CLEFT-Q scores iteratively, using progressively fewer items from the full-length PROM. Agreement between full-length CLEFT-Q score and CAT score at different assessment lengths was measured using the Pearson correlation coefficient, root-mean-square error (RMSE), and 95% limits of agreement. CAT settings, including the number of items to be included in the final assessments, were determined in a multistakeholder workshop that included patients and health care professionals. A user interface was developed for the platform, and it was prospectively piloted in the United Kingdom and the Netherlands. Interviews were conducted with 6 patients and 4 clinicians to explore end-user experience. RESULTS: The length of all 8 CLEFT-Q scales in the International Consortium for Health Outcomes Measurement (ICHOM) Standard Set combined was reduced from 76 to 59 items, and at this length, CAT assessments reproduced full-length CLEFT-Q scores accurately (with correlations between full-length CLEFT-Q score and CAT score exceeding 0.97, and the RMSE ranging from 2 to 5 out of 100). Workshop stakeholders considered this the optimal balance between accuracy and assessment burden. The platform was perceived to improve clinical communication and facilitate shared decision-making. CONCLUSIONS: Our platform is likely to facilitate routine CLEFT-Q uptake, and this may have a positive impact on clinical care. Our free source code enables other researchers to rapidly and economically reproduce this work for other PROMs.

Factors Associated With Psychological Adjustment in Adults With Cleft Lip and/or Palate: Findings From a National Survey in the United Kingdom.

OBJECTIVES: Research has identified adults born with cleft lip and/or palate (CL/P) to be at risk of poorer psychological outcomes compared to the general population. This study investigated factors that may contribute to positive and negative adjustment in adults born with CL/P. DESIGN: A survey was designed and distributed by the Cleft Lip and Palate Association in collaboration with the Centre for Appearance Research CAR at the University of the West of England (UWE). There were 207 eligible responses (95% completed online) received between July and October 2018. Dependent variables included the Body Esteem Scale for Adolescents and Adults, Harter's Self Perception Profile for Adults (Global Self-Worth, Social Competence, and Intimacy subscales), the Fear of Negative Appearance Evaluation Scale, and the Revised Adult Attachment Scale. Independent variables were the Revised Life Orientation Test, biodemographic data, and self-reported single-item questions. RESULTS: Factors associated with positive adjustment included reports of a happy childhood, talking about CL/P with family, close friendships, comfort in public spaces, satisfaction with appearance, and a positive life orientation. Psychological distress was associated with a desire for further surgery to improve appearance and/or function. CONCLUSIONS: Several factors were identified that may influence psychological adjustment in adults with CL/P. Throughout childhood, family-centered practice to support family cohesion and an open dialogue about CL/P is indicated, as is support for young people to develop social confidence. For adults returning to the cleft service, treatment options for appearance and/or functional concerns should be explored, with access to psychological support when indicated. Interventions to increase optimism, resilience, and self-acceptance may also be warranted throughout the life span.

Long-term treatment outcomes from the perspective of a patient with unilateral cleft lip and palate.

The management of patients with orofacial cleft (OFC) often extends from diagnosis or birth well into adulthood and requires many different specialists within multidisciplinary teams (MDT). The aims of treatment are to restore form and function relating to hearing, speech, occlusion and facial aesthetics. People with OFCs that include the lip, alveolus and palate (cleft lip and palate (CLP)) require several different staged and coordinated surgical and non-surgical interventions, and the treatment pathway is associated with a heavy burden of care. Due to the extensive nature of the interaction with these patients, MDT members have opportunities to provide enhanced patient-centred care and support. This case report provides an overview of the current knowledge of the aetiology of OFC and the management of these patients. It provides a unique perspective from one of the coauthors who has a unilateral CLP (UCLP) and reports on his treatment experiences and long-term treatment outcomes. By having a better understanding of the impact of UCLP and treatment provided, MDT members can not only provide improved clinical treatment but also offer improved patient experiences for those with craniofacial anomalies, in particular, an increased awareness of the psychosocial challenges, they endure throughout their treatment pathway and beyond.

Treatment Experiences in Adults Born With Cleft Lip and/or Palate: A Whole of Life Survey in the United Kingdom.

OBJECTIVES: Adults with cleft lip and/or palate (CL/P) may report dissatisfaction with aesthetic and/or functional outcomes and express desire for further treatment. Although medical intervention can improve quality of life, surgical procedures require complex decision-making and can invoke psychological distress. The aim of this study was to gain a better understanding of ongoing treatment-related support needs by examining self-reported treatment experiences of a sample of adults born with CL/P. DESIGN: An online, mixed-methods survey was designed by the Cleft Lip and Palate Association in collaboration with the Centre for Appearance Research at the University of the West of England. A total of 207 eligible responses were received. Qualitative data were analyzed using inductive content analysis, while quantitative data were analyzed using descriptive statistics. RESULTS: Although participants were satisfied with surgical and dental/orthodontic outcomes overall, many reported ongoing difficulties. Some stated they would seek further treatment while others felt conflicted about risks and benefits. Some had refused treatment, citing treatment fatigue, anxiety, and/or concerns that treatment would be unsuccessful. More than 40% of participants were unaware of their entitlement to cleft-related treatment provided by the National Health Service, and many others had experienced difficulties accessing care. CONCLUSIONS: Although advances in health care may offer opportunities for adults to improve their quality of life, treatment decisions should be weighed with the support of a clinical psychologist where available. Guidance for young adults considering future treatment is also recommended. Finally, training and resources for local practitioners may improve general awareness of CL/P services and increase access to specialist care for adults with ongoing CL/P-related concerns.