A Conceptual Thematic Framework of Psychological Adjustment in Caregivers of Children with Craniofacial Microsomia.

OBJECTIVE: Children with craniofacial microsomia (CFM) have complex healthcare needs, resulting in evaluations and interventions from infancy onward. Yet, little is understood about families' treatment experiences or the impact of CFM on caregivers' well-being. To address this gap, the NIH-funded 'Craniofacial microsomia: Accelerating Research and Education (CARE)' program sought to develop a conceptual thematic framework of caregiver adjustment to CFM. DESIGN: Caregivers reported on their child's medical and surgical history. Narrative interviews were conducted with US caregivers (n = 62) of children aged 3-17 years with CFM. Transcripts were inductively coded and final themes and subthemes were identified. RESULTS: Components of the framework included: 1) Diagnostic Experiences, including pregnancy and birth, initial emotional responses, communication about the diagnosis by healthcare providers, and information-seeking behaviors; 2) Child Health and Healthcare Experiences, including feeding, the child's physical health, burden of care, medical decision-making, surgical experiences, and the perceived quality of care; 3) Child Development, including cognition and behavior, educational provision, social experiences, and emotional well-being; and 4) Family Functioning, including parental well-being, relationships, coping strategies, and personal growth. Participants also identified a series of "high" and "low" points throughout their journey and shared their priorities for future research. CONCLUSIONS: Narrative interviews provided rich insight into caregivers' experiences of having a child with CFM and enabled the development of a conceptual thematic framework to guide clinical care and future research. Information gathered from this study demonstrates the need to incorporate evidence-based psychological support for families into the CFM pathway from birth onward.

"It's On Your Shoulders Now" Transitioning from Child-to-Adult UK Cleft Lip/Palate Services: An Exploration of Young Adults' Narratives.

OBJECTIVES: Treatment for cleft lip and/or palate (CL/P) in the United Kingdom is administered on a standardised pathway from diagnosis to early adulthood, with options to be re-referred in later life. At age 16, patients become responsible for their treatment decisions. Evidence from the wider health literature indicates this transition can be challenging and that this population may require additional support. The present study explored young people's experiences of transition to adult care in the context of CL/P services, with the aim of identifying support needs and informing future service delivery. DESIGN: Individual semi-structured interviews were conducted with 15 individuals with CL/P (aged 17-25 years) to explore transition experiences. Interviews lasted an average of 69 min and data were analysed using reflexive thematic analysis. RESULTS: Four themes, with subthemes, were identified: 1) Readiness for Transition covered feelings of preparedness and how health professionals approached transition; 2) Making Decisions as an Adult described concerns and considerations when making treatment decisions; 3) Finding and Using Support, reflected the roles of caregivers and peers in developing self-advocacy; and 4) Reflections on Transition Care offered insight into how care could be improved. CONCLUSION: Individuals born with CL/P may experience challenges in becoming responsible for their own care and treatment decisions. The findings of this study indicate that a dedicated transition protocol may be beneficial, such that adolescents are prepared to confidently access and manage their care into adulthood. Opportunities for improvements in transition planning and provision are discussed.

Early Experiences of Parents of Children With Craniofacial Microsomia.

OBJECTIVE: To describe the early health care experiences of parents of children with craniofacial microsomia (CFM), a congenital diagnosis often identified at birth. DESIGN: Qualitative descriptive. SETTING: Homes of participants. PARTICIPANTS: Parents of 28 children with CFM from across the United States. METHODS: We interviewed participants (27 mothers individually and one mother and father together) via telephone or teleconference and used reflexive thematic analysis to derive themes that represented early health care experiences of parents of children with CFM. RESULTS: Participants' narratives included detailed recounting of their birth and early care experiences. We identified two overarching themes. The first overarching theme, Stressors, included four subthemes that represented difficulties related to emotional reactions and negative experiences with health care providers. The second overarching theme, Finding Strength, included four subthemes that represented participants' positive adjustment to stressors through independent information seeking about CFM, adaptive coping, positive experiences with health care providers, and drawing on external supports. CONCLUSION: Participants often described early experiences as challenging. Findings have implications for improving early care, including increasing open and supportive communication by health care professionals, expanding access to CFM information, screening for mental health concerns among parents, strengthening coping among parents, and linking families to resources such as reliable online CFM information and early intervention programs.

Social Media and Website Use: The Experiences of Parents and Carers Accessing Care at the Oxford Craniofacial Unit.

INTRODUCTION: Historically, medical professionals have been the providers of specialist information about rare medical conditions. Now, increasingly, patients and the public are using the internet to access and generate information about medical diagnoses. The global nature of the internet allows patients to connect across geographical borders, and to obtain and share information that would have been previously inaccessible to them. This research investigated the use of website and social media by parents of children with craniosynostosis. METHODS: A cross-sectional survey-based design was employed. Participants were parents of children with craniosynostosis attending multidisciplinary craniofacial clinics within the Oxford Craniofacial Unit. A questionnaire was administered which assessed social media and website use adapted from the questionnaire created by Khouri and colleagues (2016) and Huggons and colleagues (2019). The surveys were administered over an 18-month period (November 2020 to May 2022). RESULTS: The final sample comprised 82 parents [70 mothers; 10 fathers; 1 sister/carer and 1 parent (mother/father unspecified)]. The children were aged 11 months-16 years of age (average age 6 y and 3 mo). Children had a variety of diagnoses: 31 sagittal, 19 metopic, 14 syndromic craniosynostosis, 8 unicoronal, 6 multisuture, 3 bicoronal, and 1 unilambdoid.Results showed that 93% (n=76/82) of parents used the internet to find out more about craniosynostosis, with 72% (n=59/82) of parents specifically using social media to find out more about craniosynostosis. The social media platforms used included: Facebook 64% (n=53/82), Instagram 24% (n=20/82), Blogs 12% (n=10/82), Twitter 4% (n= 4/82), Tik Tok 2% (n=2/82), and Snapchat 0.01% (n=1/82).Parents reported that Facebook was the most helpful source of information about craniosynostosis (52%; n=43/82). Parents indicated the key timepoints they used social media included: when their child received a diagnosis (70%; n=58/82), before their child's surgery (34%; n=28/82), before their first craniofacial clinic appointment (83%; n=25/30), and when child was older (17%; n=14/84). Forty percent (n=33/82) of parents said that a diagnosis of craniosynostosis made no difference to their social media use, whereas 34% (n=28/82) of parents used social media more, and 20% (n=16/82) used it less. CONCLUSION: Results highlight that parents use social media and other websites to access information relating to craniosynostosis. Future research should examine whether parental use of social media changes across their child's lifespan and evaluate the quality of this information.

Clinical Psychology and Voice Disorders: A Meta-Analytic Review of Studies Assessing Psychological Characteristics Across Individuals With and Without Voice Disorders.

OBJECTIVES: Clinical voice disorders are heterogenous conditions capturing problems with voice production and control. Psychological conceptualizations of voice disorders posit that mood, anxiety, and personality characteristics contribute to the development and maintenance of voice symptoms. This review brings together research comparing these psychological characteristics across groups with and without voice disorders, with the aim of profiling group differences. METHODS: A systematic search of PubMed, CINAHL, Ovid (PsycInfo, MEDLINE, Embase), and Web of Science databases was conducted, with studies required to assess psychological characteristics between samples with and without voice disorders. Relative study quality and risk of bias were formally evaluated, synthesizing results via meta-analysis (estimating standardized mean difference; SMD) and narrative synthesis. RESULTS: Thirty-nine studies (N = 4740) were reviewed. Marked psychological differences were observed between case-control groups, including significantly higher self-reported features of depression (SMD = 0.50), state anxiety (SMD = 0.58), trait anxiety (SMD = 0.52), health anxiety (SMD = 0.57), and neuroticism (SMD = 0.47) in voice disorder groups. However, less consistent patterns of difference were observed between voice disorder types, including minimal quantitative differences between functional and organic diagnoses. CONCLUSIONS: Findings underline and formulate the psychological features associated with experiencing a voice disorder, indicating individuals with voice disorders present with considerable psychological needs that may benefit from clinical psychology input.

The role of the clinical psychologist within a cleft service.

Clinical psychologists are core members of UK cleft services. This paper outlines the variety of ways in which clinical psychologists work across the lifespan to promote the psychological wellbeing of those born with a cleft and their families. In the context of dental or orthodontic treatment, this involves a combination of early intervention and advice, psychological assessment or specialist psychological therapy for individuals experiencing dental anxiety or anxiety regarding the appearance of their teeth. This paper therefore aims to highlight the varying roles which clinical psychologists play in the provision of cleft-related dental care, often alongside multidisciplinary colleagues.

Isolation, Uncertainty and Treatment Delays: Parents' Experiences of Having a Baby with Cleft Lip/Palate During the Covid-19 Pandemic.

OBJECTIVES: Previous literature finds that having a child with a cleft lip and/or palate (CL/P) may pose social and emotional challenges for parents. For parents of children born during the Covid-19 pandemic, such challenges may be heightened. Further, novel demands brought about by the pandemic could have caused additional hardships. The aim of this study was to describe the impact of the pandemic on new parents through qualitative exploration of their experiences. DESIGN: Semi-structured interviews were conducted with 14 parents of children born in the United Kingdom with CL/P between January and June 2020, around the start of the pandemic. Data were analysed using inductive thematic analysis. RESULTS: Three themes, with sub-themes, were identified. The first theme, "Changes to Healthcare: The Impact of Restrictions and Reduced Contact", discussed the impact of the pandemic on perinatal care, the care received from the specialist CL/P teams, and parents' experiences of virtual consultations. The second theme, "Family Functioning During the Pandemic", covered parental anxiety, fathers' experiences, and social support. The third theme, "Surgical Prioritisation: Delays and Uncertainty", addressed changes to surgical protocols, coping with uncertainty, complications associated with delayed surgery, and how parents created positive meaning from this period. CONCLUSIONS: A range of increased and additional psychosocial impacts for parents were identified, along with several coping strategies, utilization of social support, and the positive aspects of their experiences. As the pandemic continues, close monitoring of families affected by CL/P remains imperative, particularly for those at risk of emotional distress.

The Impact of the Covid-19 Pandemic on Cleft Lip and Palate Service Delivery for New Families in the United Kingdom: Medical and Community Service Provider Perspectives.

OBJECTIVES: Professionals in the United Kingdom providing care to new families affected by cleft lip and/or palate (CL/P) had to adapt to ensure families' needs were met during a time of uncertainty due to Covid-19. The aims of this study were to explore the impacts of the pandemic on CL/P care provision for new families from the perspectives of professionals working in medical and community settings along with any personal impact on professionals and their reflections on the future of CL/P care. DESIGN: Semistructured interviews (n = 27) were completed about experiences from March 2020 to October 2020 with consultant cleft surgeons (n = 15), lead clinical nurse specialists (n = 8), and staff working at the Cleft Lip and Palate Association (n = 4). Transcripts were analyzed using inductive thematic analysis. RESULTS: Three themes were identified: (1) the impact of Covid-19 on the provision of cleft care in the United Kingdom, including working conditions, delays to treatment, and Covid-19 policies; (2) the impact of the pandemic on professionals' mental health, including personal distress and concerns about Covid-19 exposure; and (3) reflections on the future of CL/P care, whereby professionals expressed both hope and concern about the Covid-19 recovery effort. CONCLUSIONS: The ongoing Covid-19 pandemic has impacted CL/P service delivery for new families significantly, warranting recommendations for cohesive psychological support for families in addition to a safe and resourced recovery effort. Support for professionals is also suggested, following existing evidence-based models for providers' needs that address the difficulties of working throughout challenging times.

Transitioning from child to adult cleft lip and palate services in the United Kingdom: Are the NICE Guidelines reflected in young adults' experiences?

Cleft lip and/or palate (CL/P) is one of the most common congenital conditions worldwide. Individuals born with CL/P will embark on a long-term treatment pathway throughout childhood and often into adulthood. As they grow older, young people become more involved in medical decisions. The National Institute for Clinical Excellence (NICE) has published guidance for health professionals on how transitions of responsibility should be managed in health services. The aim of the current study was to examine the extent to which the NICE recommendations are currently being implemented in UK CL/P services according to young adults' first-hand accounts. Semi-structured interviews were carried out with 15 young adults with CL/P aged 16-25 years. Interview questions were designed to map onto the NICE guidance. Data were analysed to assess whether each guideline was met, partially met, or not met for each individual participant. Overall, findings suggest that further consideration is needed as to how best to implement the recommendations effectively. The introduction of assigned transition workers in CL/P services to co-ordinate transition to adult care offers one possible solution. Focusing on the provision of holistic, patient-centred care, this aspect of the CL/P service could include giving patients access to medical history documentation, liaison with key health professionals including GPs and dental practitioners, and the development of age-appropriate resources to facilitate the transition process.

Staff support during COVID-19 within an Acute Hospital Trust.

BACKGROUND: The COVID-19 pandemic has put immense pressure on the National Health Service (NHS) and all healthcare professionals, not only physically but also mentally and the need to fully develop and implement a strategy to protect the mental health of healthcare professionals has never been more urgent. Research has demonstrated that staff can be supported by strengthening teams and offering frequent well-being support. AIMS: To assess the feedback from delivery of a strategy to provide psychological well-being support to NHS staff during the COVID-19 pandemic and whether this 'Support for Teams' initiative made a positive impact on staff and in what way. METHODS: A mixed methods design was used to gain quantitative and qualitative feedback from staff. Feedback was collected from two groups: Well-being Leads and clinicians providing support and resources to Well-being Leads. Feedback was collected via online forms. RESULTS: Collectively, feedback responses were received from 70 staff members. The majority of staff members felt supported and benefitted from the provisions provided. CONCLUSIONS: This evaluation showed that our healthcare system must continue to proactively implement and adapt staff support strategies to protect the mental well-being of healthcare professionals, both in the context of future health crises and in everyday practice. This study will assist and guide development and adaptations to health and psychological well-being support, ahead of future pandemics and to provide suitable support beyond the COVID-19 pandemic.

Surgical Performance Anxiety and Wellbeing Among Surgeons: A Cross-sectional Study in the United Kingdom.

OBJECTIVE: This national cross-sectional study aims to establish the prevalence and potential impact of performance anxiety among surgeons and investigate its association with psychological traits and wellbeing. SUMMARY AND BACKGROUND DATA: Despite a growing awareness that human factors, non-technical skills and wellbeing in healthcare affect patient outcomes, an area that has remained unexplored is surgical performance anxiety (SPA). METHODS: A prospectively registered, cross-sectional study using mixed methods was conducted across the United Kingdom. Data captured included demographics, surgical specialty, trait anxiety, trait perfectionism, SPA, and surgical perfectionism scores. Wellbeing was assessed using The Short Warwick-Edinburgh Mental Wellbeing Scale, whereas qualitative data were collected regarding surgeons' experiences of SPA. RESULTS: A total of 631 responses were collected. Mean age was 41·2 years and mean surgical experience 15·3 years. A total of 62% were male and 52% of consultant/attending grade. A total of 100% felt that SPA affected surgeons, with 87% having experienced it themselves. A total of 65% reported SPA negatively impacted surgical performance and 96% felt SPA negatively impacted surgeons' wellbeing. Male surgeons reported significantly better wellbeing than female surgeons. Surgeons with SPA reported significantly worse wellbeing compared with surgeons who did not experience SPA. Surgeons in general experienced significantly lower mental wellbeing compared with population norms. Thematic analysis highlighted a reticence to share SPA openly and need for cultural change. CONCLUSIONS: Surgical performance anxiety is a very common and significant challenge among surgeons across all specialties at all levels of experience in the United Kingdom. It is perceived by surgeons to affect surgical performance adversely and is associated with worse psychological wellbeing. A more open culture of sharing and acknowledgment has been identified to be beneficial.

Investigating the presence of peer bias against ptosis in young children.

PURPOSE: To investigate the presence of peer bias directed at young children aged 3-6 years of age and the age at which this bias emerges. METHODS: Children (3-6 years) completed an individual testing session involving two tasks: (1) a paired photograph task examined the effect of the appearance of ptosis on children's preferences in two different social situations (toy sharing and birthday party); (2) a ranking task investigated whether different grades of ptosis are evaluated differently by children of different ages. RESULTS: A total of 217 children were included. The paired photograph task showed that with increasing age, subjects were less likely to invite children with ptosis to share a toy or attend their birthday party. Three-year-olds did not show a bias against peers with ptosis; however, 4- to 6-year-olds did chose significantly fewer children with an apparent ptosis across both social situations. The ranking task highlighted that 6-year-olds viewed a grade 3 ptosis significantly more negatively than 3- and 4-year-olds; however, there was no effect of age on the rankings given to other grades of ptosis. CONCLUSIONS: Bias against ptosis emerges at about 4 years of age and is still evident at 6 years.

Neurodevelopmental, Cognitive, and Psychosocial Outcomes for Individuals With Pathogenic Variants in the TCF12 Gene and Associated Craniosynostosis.

ABSTRACT: Heterozygous mutations in the TCF12 gene were discovered in 2013 as a cause of craniosynostosis (CS). However, limited information regarding the behavioral phenotypic profile is available. Here the authors provide the first detailed study of the neurodevelopmental, cognitive, and psychosocial outcomes for patients with a pathogenic TCF12 variant and associated CS.A clinical casenote audit was conducted at the 4 UK highly specialized craniofacial centers. A total of 35 patients aged 18 months to 10 years with an identified TCF12 pathogenic variant and CS (bicoronal CS = 45.7%, unicoronal CS = 40.0%, multisuture = 14.3%) were included. Standardized screening and/or assessment of full-scale intelligence quotient, social communication, development, behavior, and self-concept were conducted.In the majority of cases, outcomes were consistent with age-related expectations. About 75% of patients demonstrated no delay across any early developmental domain, while 84.6% demonstrated full-scale intelligence quotient scores within 1 standard deviation of the population mean. Significant behavioral difficulties were demonstrated by parent reporters in 26.3% to 42.1% of cases (dependent upon domain). Clinically elevated social communication profiles were present in (41.7%) of parent-reported cases. Levels of self-concept (at age 10) were consistent with age-related normative data.Most patients with a TCF12 pathogenic variant had a mild behavioral and cognitive phenotype, although they may be at a slightly increased risk of social communication difficulties and psychosocial issues. Although not measured statistically, there were no clear associations between surgical history and cognitive, behavioral, or psychosocial outcomes. This paper highlights the need for robust integrated developmental assessment of all CS patients, particularly those with an identified syndrome.

'Your face freezes and so does your life': A qualitative exploration of adults' psychosocial experiences of living with acquired facial palsy.

OBJECTIVES: Facial palsy (FP) is a highly visible appearance-affecting condition and can have a significant impact on facial function. Qualitative research focussing on adults' experiences of living with acquired FP is limited. This study aimed to explore the psychosocial impact of acquired FP and to gain a greater understanding of patients' experiences of treatment and care in the United Kingdom. DESIGN: A qualitative interview study with individuals living with acquired FP. METHODS: Ten adults with acquired FP were recruited. Their experiences were explored using semi-structured telephone interviews. Data were analysed using thematic analysis. RESULTS: Five master themes were identified through the thematic analysis: 1) grappling with a new identity, 2) the psychosocial impact of living with facial palsy, 3) isolation: dealing with 'one hell of a problem on your own', 4) a life on hold, 5) coping strategies. Findings indicated high levels of distress and significant challenges in managing the functional and psychosocial changes associated with acquiring FP. Participants expressed grief for their former appearance and identity, with photographs and mirrors acting as agonizing reminders. Many reported a sense of abandonment due to uncoordinated care and, as a result, engaged in an endless and often fruitless pursuit to gain control over FP by experimenting with their own treatment. Many reported the negative impact of their altered facial expressions on social interactions and a fear of being negatively evaluated. CONCLUSIONS: This study highlights a pressing need to review how FP is managed in the UK. To improve patient well-being, health care professionals could benefit from FP education, and patients from timely access to psychological support and clearer standards of care following diagnosis.

A Systematic Review of the Psychosocial Adjustment of Children and Adolescents with Facial Palsy: The Impact of Moebius Syndrome.

INTRODUCTION: Facial palsy is often associated with impaired facial function and altered appearance. However, the literature with regards to the psychological adjustment of children and adolescents with facial palsy has not been systematically reviewed to date. This paper aimed to review all published research with regards to psychosocial adjustment for children and adolescents with facial palsy. METHODS: MEDLINE, CINAHL, Embase, PsychInfo and AMED databases were searched and data was extracted with regards to participant characteristics, study methodology, outcome measures used, psychosocial adjustment and study quality. RESULTS: Five studies were eligible for inclusion, all of which investigated psychosocial adjustment in participants with Moebius syndrome, a form of congenital facial palsy. Many parents reported their children to have greater social difficulties than general population norms, with difficulties potentially increasing with age. Other areas of psychosocial adjustment, including behaviour, anxiety and depression, were found to be more comparable to the general population. DISCUSSION: Children and adolescents with Moebius syndrome may experience social difficulties. However, they also demonstrate areas of resilience. Further research including individuals with facial palsy of other aetiologies is required in order to determine the psychosocial adjustment of children and adolescents with facial palsy.

The psychosocial impact of facial palsy: A systematic review.

PURPOSE: Facial palsy is a condition which can lead to significant changes in facial function and appearance. People with facial palsy often report psychosocial difficulties, including withdrawal from social activities, anxiety, negative body image, and low mood. This paper aimed to review all published research investigating the psychosocial impact of facial palsy on adults. METHODS: A systematic search of MEDLINE, CINAHL, EMBASE, PsycINFO, and AMED databases was performed. The quality of included studies was assessed, and data were extracted with regard to characteristics of participants; study methodology and design; outcome measures used; and psychosocial outcomes. RESULTS: Twenty-seven studies met inclusion criteria. A high proportion of people with facial palsy reported clinically significant levels of anxiety and depression, with greater difficulties typically reported by females, compared to males. Other difficulties consistently reported include low quality of life, poor social function, and high levels of appearance-related distress. Objective severity of facial palsy was consistently shown to not be associated with anxiety or depression, with psychological factors instead likely mediating the relationship between the severity of facial palsy and psychosocial well-being. CONCLUSIONS: Irrespective of objective symptom severity, facial palsy has the potential to have a significant impact on psychosocial well-being and quality of life. The various methodological limitations of the included studies are discussed, along with clinical implications, including the need for greater access to psychological screening and interventions for people with facial palsy.

The Psychosocial Adjustment of Children Born With a Cleft Lip and/or Palate: Cross-Sectional and Longitudinal Analyses.

OBJECTIVE: To determine the psychosocial adjustment of children born with a cleft lip and/or palate (CL/P). DESIGN: Longitudinal analyses of psychosocial outcomes and cross-sectional comparison with published norms. SETTING: Retrospective clinical audit at a UK cleft center. PARTICIPANTS: Data available for 1174 participants born with a CL/P at ages 5 (n = 658), 10 (n = 415), and 15 (n = 171), with longitudinal data for a subset of the sample at 5 and 10 (n = 168) and 10 and 15 (n = 49). MAIN OUTCOME MEASURE: Parental-report Strengths and Difficulties Questionnaire (SDQ). RESULTS: The majority of children scored in the average range for overall adjustment. Children born with CL/P had significantly higher mean overall SDQ scores relative to normative data at ages 5 and 10. Longitudinal analysis highlighted that adjustment at age 5 was a significant predictor of adjustment at age 10. Gender was another significant predictor of adjustment at age 5 and 10, with boys reporting more problems than girls. However, effect sizes for the impact of age and gender were small. Cleft type was not a significant predictor of SDQ score at any age. CONCLUSIONS: Children aged 5 and 10 years of age born with a cleft may experience greater overall psychosocial difficulties than the general population. The domains on the SDQ on which children experience difficulty may be influenced by age and gender. Future research should focus on the specific impact of cleft-related issues, including speech, language, and hearing difficulties, on psychosocial adjustment.

A systematic review of the use of psychological assessment tools in congenital upper limb anomaly management.

STUDY DESIGN: This study is a systematic review. INTRODUCTION: Congenital upper limb anomalies (CULAs) are often associated with psychosocial difficulties including negative body image, low self-esteem, and withdrawal from social activities. PURPOSE OF THE STUDY: The purpose of the study was to identify, describe, and evaluate all published psychosocial assessment tools used in the assessment and management of CULAs, to direct the use of these tools in clinical practice, and to identify areas requiring development. METHODS: A systematic search of Medline, EMBASE, Pubmed, and PsychInfo databases was performed. In total, 23 studies were included for analysis. Data extracted included study and population characteristics, psychosocial measures utilized, psychosocial outcomes reported, and the reliability and validity of measures. RESULTS: Seventeen patient-reported measures were identified. The most commonly used tool was a Likert scale (n =7) with satisfaction with appearance and function, the most commonly evaluated outcome (n = 18). Other evaluated domains included quality of life or psychosocial functioning (n = 9), self-image (n = 2), and psychological well-being (n = 5). DISCUSSION AND CONCLUSIONS: There is no well-established, validated assessment tool in regular use to effectively address psychosocial outcomes for children with CULAs. Although the majority of children born with a CULA appear to adjust well, this is by no means the case for all children. There is a need for routine psychosocial evaluation preoperatively and postoperatively with long-term follow-up data to help direct patient-orientated management. A clear understanding of these, and how to measure them, is needed to help for a patient-centred, multidisciplinary, evidence-driven approach to CULA management.