Emotional Wellbeing in Adults Born with Cleft Lip and/or Palate: A Whole of Life Survey in the United Kingdom
Authors: Kenny Ardouin, Jess Hare, Nicola Stock
Journal: Cleft Palate-Craniofacial Journal
Abstract
Objectives: Previous research with adults born with cleft lip and/or palate (CL/P) has identified a range of concerns regarding mental health and quality of life, concluding that overall emotional functioning is poorer compared to the general population. The aim of the current study was to build on this previous work by investigating the self-reported emotional well-being of adults born with CL/P in the United Kingdom.
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 between July and October 2018. Qualitative data were analyzed using inductive content analysis, while quantitative data were analyzed using descriptive statistics and independent t tests.
Results: Almost half of the sample reported a diagnosis of a mental health condition. Scores on standardized measures indicated significantly lower global self-worth, higher levels of fear of negative appearance evaluation, and lower self-perceived job competence compared to normative data. Scores of overall body esteem were in line with the general population. Most participants had not received psychological support from their regional CL/P team and 41% were unaware of their entitlement to treatment from the National Health Service.
Conclusions: Individuals with CL/P may be at risk of emotional distress persisting into adulthood. The integration of routine psychological support from an early age is highly recommended, as is information for young adults about to complete routine treatment. Comprehensive psychological screening for adults returning to the service later in life is also strongly advised.
Physical Health in Adults Born With Cleft Lip and/or Palate: A Whole of Life Survey in the United Kingdom
Authors: Kenny Ardouin, Sinead Davis, Nicola Stock
Journal: Cleft Palate-Craniofacial Journal
Abstract
Objectives: Previous research in the field of cleft lip and/or palate (CL/P) has indicated a high prevalence of common health concerns, functional difficulties, and additional conditions known to affect long-term physical health, cognitive development, and psychological well-being. The aim of the present study was to examine the self-reported physical health of a national sample of adults born with CL/P.
Design: Drawing upon previous literature, 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 between July and October 2018. Qualitative data were analyzed using inductive content analysis, while quantitative data were analyzed using descriptive statistics.
Results: A considerable proportion of participants reported one or more co-morbid conditions, including common health concerns, difficulties related to language and/or learning, unresolved speech and/or hearing issues, problems with vision, breathing difficulties, and concerns related to eating and drinking. Many participants were unaware of their entitlement to specialist treatment, and some had experienced difficulties accessing specialist services through their general practitioner (GP).
Conclusions: Individuals with CL/P may be at risk of physical health issues persisting into adulthood. The monitoring of physical symptoms from an early age is recommended, as is a routine physical assessment for adults returning to the CL/P service later in life. Education for both GPs and adults is warranted. Future studies could investigate the wider prevalence of co-morbid conditions in CL/P, in order to better understand the longer-term health burden.
Interpersonal Relationship Experiences in Adults Born With Cleft Lip and/or Palate: A Whole of Life Survey in the United Kingdom
Authors: Kenny Ardouin, Matt Hotton, Nicola Stock
Journal: Cleft Palate-Craniofacial Journal
Abstract
Objectives: Previous research has suggested that adults with a cleft lip and/or palate (CL/P) may find their familial, friend, collegial, and/or romantic relationships impacted by their condition. Building on this prior work, this study examined the self-reported interpersonal experiences of a national sample of adults born with CL/P.
Design: An online survey was designed by the Cleft Lip and Palate Association in collaboration with the Centre for Appearance Research. Data from 181 participants were included in the present study, collected between July and October 2018. Qualitative data were analysed using inductive content analysis, while quantitative data were analyzed using descriptive statistics and independent t tests.
Results: Most participants reported having good relationships with family members and existing friends with similar patterns of marriage as the general population. However, some experienced difficulties initiating new friendships and romantic relationships. Participants described bullying and discrimination in adulthood, most often in public settings and the workplace. Participants reported lower satisfaction with intimate relationships and less secure attachment compared to the general population. Concerns regarding the heritability of CL/P and its impact on the decision to have a family were also reported.
Conclusions: Individuals with CL/P are at risk of negative social experiences, which if not appropriately addressed may impact psychological well-being in adulthood. The findings illustrate the importance of routine psychosocial support from childhood onward to help individuals with CL/P build social skills, self-esteem, and social confidence and to develop mutually fulfilling friendships and intimate relationships. Additionally, societal campaigns to raise awareness and target discrimination may be helpful.
Treatment Experiences in Adults Born With Cleft Lip and/or Palate: A Whole of Life Survey in the United Kingdom
Authors: Kenny Ardouin, David Drake, Sandip Popat, Nicola Stock
Journal: Cleft Palate-Craniofacial Journal
Abstract
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 analysed 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.
Factors Associated With Psychological Adjustment in Adults With Cleft Lip and/or Palate: Findings From a National Survey in the United Kingdom
Authors: Bruna Costa, Kenny Ardouin, Nicola Stock
Journal: Cleft Palate-Craniofacial Journal
Abstract
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 University of the West of England. 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.
Expanding Support Services for Adults Born With Cleft Lip and/or Palate in the United Kingdom: An Exploratory Evaluation of the Cleft Lip and Palate Association Adult Services Programme
Authors: Kenny Ardouin, Nicky Davis, Nicola Stock
Journal: Cleft Palate-Craniofacial Journal
Abstract
Background: The largest group of people living with repaired cleft lip and/or palate (CL/P) are adults. Previous research has identified unmet treatment and psychosocial needs, yet few interventions exist. This article outlines 3 interventions developed by the Cleft Lip and Palate Association as part of its 3-year community-based Adult Services Programme; an Adults Conference, a series of panel discussions (“Cleft Talk”) streamed in podcast/video format, and a Leaver’s Pack of resources for adults wishing to
return to cleft care.
Methods: Feedback from attendees of the Adults Conferences (2018-2019) was collected using specifically developed evaluation forms. Streaming metrics and social media interactions were extracted for Cleft Talk panel discussions (2019-2020). The Leaver’s Pack was piloted in 2020, using an online evaluation form. Specialist health professionals were invited to provide feedback or
participate in a one-to-one interview regarding their perceived impact of the program.
Results: All 3 interventions across the different modalities received support from participating adults, demonstrating potential to meet adults’ needs across the life span. Health professionals also offered support for the program, viewing the interventions as a valuable adjunct to formal medical CL/P services.
Conclusions: This exploratory evaluation indicates that peer- and community-led interventions, in combination with ongoing access to specialist medical care, can have a range of positive impacts for adults with CL/P. There is scope for similar initiatives to be developed internationally and for individuals with other craniofacial conditions. Not-for-profit organizations are encouraged to
routinely evaluate their interventions to create a stronger evidence base for their valuable work.
Orthognathic Surgery: My Experience of Transitioning to Adult Cleft Care
Author: Kenny Ardouin
Journal: Cleft Palate-Craniofacial Journal
Abstract
Kenny Ardouin grew up in East Sussex in the United Kingdom (UK), before moving to New Zealand, which he now considers home. Having been born with a cleft lip and palate, Kenny has a personal and professional dedication to the field. He served as CEO of the charitable organization Cleft New Zealand and recently completed 3.5 years as the Adult Services Manager for the Cleft Lip and Palate Association in the UK. Kenny works as a speech and language therapist, and is studying a Master’s degree part-time, focused on the psychological impact of speech differences in adolescents and adults born with a cleft. Kenny is also a radio broadcaster and editor and a freelance public speaker. In this heartfelt and informative invited editorial, Kenny reflects on how his treatment journey unfolded as a young adult and offers key learning points for all health professionals wanting to become more person-focused in their clinical practice.
Long-term treatment outcomes from the perspective of a patient with unilateral cleft lip and palate
Authors: Peter Fowler, Kenny Ardouin, Jennifer Haworth, Leslie Snape
Journal: British Medical Journal Case Reports
Abstract
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.
Understanding the long-term effect of speech differences in cleft lip and palate: a study of how cleft-related speech differences influence educational, vocational and social experiences in New Zealand adults.
Authors: Kenny Ardouin
Thesis: MSc Thesis, University of Canterbury
Abstract
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.
Background: The largest group of people living with repaired cleft lip and/or palate are adults. Previous research has identified that cleft-related differences and concerns can persist well into adulthood, and cleft is therefore gaining increasing recognition as a lifelong condition. Although the physiological impact of cleft on speech is well understood, the social and psychological impacts of cleft-related speech differences are less explored. The aim of this study was to examine self-reported perceptions of cleft-related speech differences in adulthood, and their educational, vocational and social impacts on New Zealand adults.
Method: Drawing upon previous international literature, a mixed-methods study was designed. This primarily involved individual semi-structured qualitative interviews with adults born with cleft. Interviews lasted between 30-60 minutes, and were followed with a form containing five quantitative standardised measures. A total of 17 participants aged between 18 and 84 participated in the study between August and November 2020. Sixteen participated using Zoom video conferencing software, and one participant opted for a face-to- face interview. Qualitative data were analysed using thematic analysis, while quantitative data were analysed using descriptive statistics.
Results: Most participants perceived themselves to have some ongoing speech concerns arising from their cleft, with participants overall reporting their speech acceptability to be poorer than their speech intelligibility. While most people identified ongoing cleft related speech concerns, a proportion of these also reported cleft related appearance concerns. Four overarching themes associated to cleft-related speech differences were identified. The first theme explored participants’ perceptions of speech differences and speech language therapy (SLT). The remaining themes focussed on the impact of cleft-related speech differences on 1) educational and vocational experiences, 2) social and interpersonal experiences, and 3) emotional wellbeing. Cleft-related speech differences impacted on participants’ educational and vocational experiences in a myriad of ways, including choosing to opt out of opportunities and being subjected to discrimination, as well as positively influencing their choice of vocation. Overall, participants reported their social competence to be lower than that of the general population despite reporting healthy relationships with friends and family. Participants typically reported establishing new friendships and romantic relationships to be difficult. Finally, emotional wellbeing concerns were prevalent in the cohort, with access to psychology support services having been limited. Conclusion: Responses from this self-selecting cohort indicated that cleft-related speech concerns and/or the psychological impact of previous or current cleft-related speech concerns may persist well into adulthood. The lack of access by some in the cohort to speech language therapy services growing up, as well as the lack of access to psychological services by almost all of the cohort are areas worthy of further attention. Recommendations for clinicians and charitable services working in cleft care are offered, in addition to recommendations for future health policy, which would standardise access to cleft care across New Zealand. Further research both locally and internationally is recommended to better understand the long-term impact of cleft-related speech differences, and to evaluate the efficacy of any interventions implemented as a result of this study.
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
Authors: Conrad Harrison, Inge Apon, Kenny Ardouin, Chris Sidey-Gibbons, Anne Klassen, Stefan Cano, Karen Wong Riff, Andrea Pusic, Sarah Versnel, Maarten Koudstaal, Alexander C Allori, Carolyn Rogers-Vizena, Marc C Swan, Dominic Furniss, Jeremy Rodrigues
Journal: Journal of Medical Internet Research
Abstract
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.
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