Objective: Assess the level of agreement for classification of orofacial clefts within and between different raters and validate classifications. Design: Validation study. Setting: National longitudinal prospective cohort, United Kingdom. Participants: Children born with orofacial cleft (n=4211), recruited to the Cleft Collective between 2013 and 2024. Main outcome measures: Four cleft classifications of orofacial clefts were explored. Classification one comprised cleft lip, cleft palate, and cleft lip and palate. Classification two added laterality, classification three added sidedness and classification four added completeness using LAHSAL. Data on orofacial cleft classification were collected through seven different sources. Results: At least one report of cleft classification was available for 4052 Cleft Collective study children. When assessing intra-rater agreement mothers had the highest level of agreement for the simplest form of cleft classification with a Krippendorf's Alpha of 0.987. When recording LAHSAL for the same child, surgeons reported the same classification for 71% of children (Krippendorf's Alpha= 0.672). When assessing inter-rater agreement across different sources, the simplest cleft classification resulted in the highest level of agreement (Krippendorf's Alpha = 0.957) and the least agreement when using LAHSAL (Krippendorf's Alpha = 0.538). Conclusion: Our study found that the more complex a cleft classification becomes the less agreement there is between sources. Differences across sources became most apparent when reporting the sidedness or completeness of a cleft. Validation of cleft classification is important for both clinical care and research. Although LAHSAL is advocated for use both clinically and in research our data show that rigorous training is essential.

Abstract Background Children with cleft lip and/or palate (CL/P) experience increased rates of neurodevelopmental difficulties, including ADHD, autism spectrum disorder, and educational challenges. While rare neurodevelopmental copy number variants (ND-CNVs) are enriched in this population and associated with poorer outcomes, these variants are present in only a small proportion of children born with cleft. Whether shared common genetic variation contributes to neurodevelopmental comorbidities in CL/P remains unknown. Methods We investigated this question using data from 2,313 children with CL/P from the Cleft Collective and 7,913 population controls from the Millennium Cohort Study. We tested for shared genetic architecture using linkage disequilibrium score regression, examined associations between polygenic risk scores for eight cognitive, neurodevelopmental, and psychiatric traits and developmental and behavioural outcomes within the cleft population, compared polygenic risk scores between cases and controls, compared polygenic burden between ND-CNV carriers and non-carriers, and employed two-sample Mendelian randomization to test whether genetic liability to cleft causally influences neurodevelopmental outcomes. Results Linkage disequilibrium score regression revealed little evidence of genetic correlations between CL/P and any of the eight traits examined. Within the cleft population, polygenic risk scores demonstrated expected associations with developmental and behavioural outcomes; however, children with CL/P did not have increased polygenic risk scores for ADHD, autism, depression, anxiety, schizophrenia, bipolar disorder, or lower scores for educational attainment or intelligence compared to controls. Mendelian randomization provided no robust evidence that genetic liability to cleft causally influences neurodevelopmental outcomes. ND-CNV carriers did not differ from non-carriers in polygenic burden. Conclusions The increased neurodevelopmental risk observed in CL/P does not appear to be primarily explained by shared common genetic architecture with psychiatric disorders, in contrast to established rare variant contributions. Polygenic risk scores for neurodevelopmental traits predict behavioural outcomes within the cleft population similarly to the general population, suggesting these genetic factors may be operating independently of cleft status while remaining clinically relevant.

Objective Cleft lip and palate significantly impact a child’s speech and facial appearance. Children undergo cleft repairs in infancy, but poor results from these initial repairs often lead to secondary surgery. In the late 1990s, cleft care provision in the UK was centralised to approximately 11 managed clinical networks or centres. This centralisation has been associated with improvements in speech and aesthetic outcomes, but little is known about the effect of centralisation on the use of secondary surgery. The purpose of this study was to compare the cumulative incidence of secondary cleft surgeries before and after centralisation and the proportion of children achieving good clinical outcomes without secondary surgery.Design Retrospective, cross-sectional.Setting and participants Two cross-sectional studies of 5-year-old children with non-syndromic unilateral cleft lip and palate were conducted, one precentralisation and one postcentralisation.Outcome measures The cumulative incidence of secondary surgery from birth through age 5 was compared precentralisation and postcentralisation using Fisher’s exact test, as were facial appearance and speech outcomes at age 5. Risk ratios (RR) were estimated using log-binomial multivariable regression models that adjusted for sex and age at evaluation.Results Postcentralisation, the proportion of children achieving good or excellent facial appearance increased from 16% to 42% (p<0.0001), good speech outcomes improved from 82% to 90% (p=0.02) and those avoiding secondary surgery rose from 45% to 67% (p<0.0001). The risk of secondary surgery decreased by 40% (RR: 0.60; 95% CI: 0.48 to 0.74), with notable reductions for secondary lip, palate and nose surgeries (RR: 0.19, 0.54 and 0.13, respectively; p<0.0001). The proportion of children achieving an ideal surgical outcome—good facial appearance, good speech and no secondary surgery—increased from 7% precentralisation to 28% postcentralisation (p=0.01; 4.1-fold increase).Conclusions Centralisation of cleft care was associated with improved outcomes of primary lip and palate repairs and a corresponding reduction in secondary surgery.

Scars resulting from surgical repair of cleft lip and palate provide a unique model in humans for studying factors that affect the severity of scarring. Anatomically, clefts of the lip and palate are consistent in location and are treated surgically with similar methods and timing. This contrasts with the considerably greater variability observed in traumatic injuries, burns or emergency surgical procedures and the scars resulting from these cause tissue damage. Clinical audit protocols for cleft lip and palate ensure that data on outcomes for appearance, function and wellbeing are collected at standard points in a child's development and are followed-up over time, permitting cross sectional and longitudinal analysis of scarring. Therefore, enhancing our understanding of the impact of genetics and environmental factors on scar severity following surgical repair of clefts can provide information on factors that may be associated with scarring from other insults, where uniform data are not available. Animal models offer opportunities to investigate factors that could influence scar severity and overall repair quality. In this review, we will discuss the possibilities for scarring research in cohorts of children born with cleft defects and the advantages of using different animal models for studying the mechanistic drivers of variation in scar severity.

Several genome wide association studies (GWASs) of orofacial cleft have been conducted. However only a few such studies to date have combined all cleft cases, focused on subtypes other than non-syndromic cleft lip with/without cleft palate, or investigated subtype heterogeneity. We conducted a GWAS of orofacial clefts within 2268 cases from the Cleft Collective and 7913 population-based controls; we performed analyses of all orofacial clefts, plus 7 subgroups. We replicated our findings in a meta-analysis of independent samples and investigated patterns of correlation across subgroups. We identified 27 regions at genome-wide significance, 8 of which were novel. We also conducted the first GWAS of Pierre Robin Sequence, despite the small sample size (n cases = 237), we found one genome wide significant SNP (P < 5 × 10-8), and another 21 suggestive associations (P < 10-5). Novel loci include those mapping to LHX8 and TSBP1 (combined clefts), ARHGEF18 and ARHGEF19 (cleft lip with/without palate), FBN2 (cleft lip only), SLC35B3 (cleft palate only), CASC20 (Pierre Robin Sequence) and CHRM2 (non-syndromic cleft palate only). Several novel hits were in regions previously associated with facial morphology in GWAS or were in regions involved in key developmental processes, including neural crest cell migration and craniofacial development. We identified genetic loci with similar effects across all subgroups and some loci which were subtype specific, we also identified 3 loci with opposing effects on cleft lip and Pierre Robin sequence. Our findings highlight the merit of including all orofacial cleft subtypes in GWAS studies and investigating heterogeneity of effects across subtypes.

To investigate the interdental arch relationship outcomes of 5-year-old children with unilateral cleft lip and palate (UCLP) before and after centralization of cleft services in the United Kingdom (UK) using the modified Huddart-Bodenham index (MHBI) and to investigate any differences in MHBI by cleft laterality. Retrospective cross-sectional study. Evaluation of three-dimensional study models of children with a complete UCLP. All available 5-year-old orthodontic study models of participants with UCLP from the precentralization Clinical Standard Advisory Group (CSAG n = 107) and postcentralization cleft care UK (CCUK n = 195) studies. The models were also grouped by cleft laterality (left and right sided). Differences between the interdental arch relationship outcomes were assessed using the MHBI. This index scored the buccal/palatal or labial/palatal relationships of 8 maxillary deciduous teeth with the opposing mandibular dentition. The anterior segment, buccal cleft segment, and noncleft segment scores were calculated and combined to calculate the MBHI total arch scores. Inter and intraexaminer reliability demonstrated high levels of agreement. Statistically significant differences in the anterior segment, buccal noncleft segment, and total arch MHBI scores were found, with postcentralization CCUK performing better. Right-sided UCLP had statistically significantly better buccal cleft segments, but no differences were found for the other MBHI segments or total arch scores. There were improved interdental arch relationships postcentralization of cleft services in the United Kingdom. Cleft laterality differences were limited to the buccal cleft segment with right-sided UCLP having better MBHI scores.

Cleft lip and/or palate (CL/P) is the most common craniofacial congenital anomaly and has been associated with higher risk of neurodevelopmental and behavioural problems indicating potential shared genetic factors between CL/P and neurodevelopmental disorders. In this study, we aimed to determine the prevalence of neurodevelopmental copy number variants (CNV) in children with CL/P and their link to early developmental and behavioural problems. Using data from the Cleft Collective, the largest UK-based national cohort study of children with CL/P, we determined the rates of neurodevelopmental CNVs in children with CL/P comparing them to the general population, explored differences by cleft type and investigated risk of developmental delays and behavioural problems among those with CL/P and neurodevelopmental CNVs. Children with CL/P had a higher prevalence of neurodevelopmental CNVs than participants in four population-based samples (3.7% vs 2.3% in the Avon Longitudinal Study of Parents and Children (ALSPAC), 2.0% in Born in Bradford (BiB), 2.3% in Millenium Cohort Study (MCS), 1.7% in UK Biobank, ORs(95%CIs): ALSPAC = 1.56(1.18–2.06), BiB = 1.84(1.37–2.45), MCS = 1.59(1.19–2.11), UK Biobank = 2.15(1.68–2.71). Children with cleft palate only were 3 times more likely to have a neurodevelopmental CNV (95%CIs1.50–6.59, p = 0.03) than children with cleft lip only. Furthermore, children with CL/P and neurodevelopmental CNVs were more likely to experience early developmental delays and behavioural problems by age 5 compared to children with CL/P and without neurodevelopmental CNVs. These findings highlight that genetic testing ascertaining the presence of neurodevelopmental CNVs might be helpful in early identification of developmental needs in children with CL/P.

Introduction Cleft lip and/or palate (CL/P) affects 1 in 700 live births globally. Children born with CL/P and their families face various challenges throughout the child’s development. Extant research is often limited by small numbers and single-centre data. The Cleft Collective, a national cohort study in the UK, aims to build a resource, available to collaborators across the globe, to understand causes, best treatments and long-term outcomes for those born with CL/P, ultimately seeking to enhance their quality of life through improved understanding and care.Methods and analysis A longitudinal prospective cohort study of children born with CL/P and their families. Recruitment occurs across the UK and started in November 2013. Recruitment will continue until September 2027 with an estimated final sample of 4822 children born with CL/P (1157 cleft lip including/excluding the alveolus; 2112 cleft palate only; 1042 unilateral cleft lip and palate and 511 bilateral cleft lip and palate). Biological samples are collected from all recruited members of the family. Parental and child questionnaires are collected at key time points throughout the child’s development. Surgical data are collected at the time of surgical repair of the child’s cleft. Consent is obtained to link to external data sources. Nested substudies can be hosted within the cohort. Regular engagement with participants takes place through birthday cards for the children, social media posts and newsletters. Patient and Public Involvement is conducted through the Cleft Lip And Palate Association and Cleft Collective Patient Consultation Group who provide insightful and essential guidance to the Cleft Collective throughout planning and conducting research.Ethics and dissemination The Cleft Collective was ethically approved by the National Research Ethics Service committee South West—Central Bristol (REC13/SW/0064). Parental informed consent is required for participation. Findings from the Cleft Collective are disseminated through peer-reviewed publications, conference presentations, newsletters and social media.

Coordinated biological responses are required to bring about orthodontic tooth movement. Several theories have been proposed for the mechanisms underlying tooth movement. Techniques have been investigated for reducing pain associated with orthodontics, as well as increasing the rate of tooth movement. Modern molecular and genetic techniques have improved our understanding of biological processes underlying tooth movement, but there is still a great deal of detail that is unknown. CPD/Clinical Relevance: This article provides an update on contemporary theories of orthodontic tooth movement.

Objective The primary aim was to assess the cost (£) to the National Health Service (NHS) of hospital treatment for individuals born with bilateral cleft lip and palate (BCLP), unilateral cleft lip and palate (UCLP), and cleft palate (CP), from birth to 20 years. The secondary aim was to assess the number of scheduled clinical outpatient and inpatient/outpatient surgical visits for this cohort. Design Retrospective micro-costing analysis. Setting Patients treated within the South West of England Cleft Service (UK). Patients Twenty-three case-notes of children born with cleft (5 BCLP; 10 UCLP; 8 CP) were identified from birth up to and including secondary alveolar bone graft (ABG), and a second group of 23 hospital case-notes (3 BCLP; 10 UCLP; 10 CP) were identified, post-ABG to 20 years. Results Mean costs from birth to ABG were £17,004 (BCLP), £11,620 (UCLP), and £6137 (CP), and post-ABG to 20 years were £9,463, £7,945, and £3,816, respectively. The largest costs were for staff. Repeat surgery had a significant impact on costs. The mean number of clinical outpatient visits for BCLP, UCLP, and CP were 140, 110 and 83 respectively and 8, 6 and 2 for inpatient/outpatient surgical visits, respectively. Conclusions Costs for provision of cleft care by the NHS are significant. The greatest costs were incurred with care for patients with BCLP. Patients were expected to attend many appointments, with BCLP experiencing the most visits. Engagement with quality improvement programmes to minimise repeat surgery, and remote consultation for certain appointments, to reduce the patient burden are recommended.

Background: Orofacial clefts are the most common craniofacial congenital malformation in humans. Approximately 30% of clefts arise as part of a syndrome or sequence, characterised by co-existing structural and functional anomalies. Many syndromes are thought to be undiagnosed, although the presence of multiple anomalies may indicate the presence of a syndrome or sequence. Aim: To determine the extent to which the presence of additional structural and functional anomalies can help to identify those children with an undiagnosed syndrome. Methods: Secondary data analysis was performed using data from 1701 children born with an orofacial cleft, collected as part of a longitudinal cohort study, the Cleft Collective. Data were collected between 2013 and 2023 across the United Kingdom. The prevalence of structural and functional anomalies and syndromes were explored using descriptive statistics. Logistic regression was used to determine the extent to which anomalies can predict syndromic status. Results: A syndrome and/or sequence was reported in 20.5% children. Among children who reported five or more anomalies, the prevalence of a diagnosed syndrome was 81.5%. When adjusting for cleft subtype and sex, in 27 out of 32 anomalies tested, strong evidence was found to suggest increased odds of having a syndrome if the specific anomaly was present compared to if the anomaly was absent (p-values ranged between 1.4 × 10−30 and 0.002). Conclusions: Children born with a cleft who present with two or more anomalies are much more likely to have a syndrome than those with fewer anomalies and should be prioritised for genetic screening and counselling.

Objectives To estimate both the association of surgical variables in complete unilateral cleft lip and palate (cUCLP) in the UK with outcomes at age 5 years, and the association of secondary speech surgery, volume of surgery, and surgeon with the same outcomes. Setting and Sample Population The Cleft Care UK study, a cross-sectional study of 268 5-year-olds, born from 2005 to 2007, with cUCLP. Materials and Methods Information on surgical variables was extracted from a standardized questionnaire. Dento-facial outcomes were derived from dental study casts of dental arch relationships. Three speech outcomes - intelligibility, structure and articulation - were derived using the Cleft Audit Protocol for Speech-Augmented tool. Results Surgical and outcome data were available for 211 (79%) children from all cleft centres in the UK. Later soft palate surgery was associated with a 17% increased chance of a poor intelligibility score (P = .02), and high volume surgery with a 249% increased chance of a good articulation score (P = .01). There were no between surgeon effects identified. No association between the surgical variables examined and dento-facial outcome, or secondary speech surgery by the age of 5 years were found. Conclusion This study found associations between surgical variables and speech outcomes at 5 years of age, but not between surgical variables and dento-facial outcome, nor between surgical variables and secondary speech surgery. High surgical volume should be maintained, and any changes towards later surgery monitored for changes in speech outcome.

Objective To assess factors affecting willingness to pay for orthodontic treatment.Methods An online discrete choice experiment and willingness to pay study was conducted on a convenience sample of 250 participants aged 16 and above over a four-month period. Participants completed a series of stated-preference tasks, in which they viewed choice sets with two orthodontic treatment options involving different combinations of attributes: family income; cost to patient; cause of problem; prevention of future problems; age; severity of the problem; and self-esteem/confidence.Results Family income, cost to patient, cause of the problem, age and self-esteem/confidence were the most important attributes influencing participants' decisions to have orthodontic treatment. Participants felt that free NHS-based orthodontic provision should be prioritised for those under 18, regardless of family income, for those with developmental anomalies, particularly where self-esteem and confidence are affected, with younger participants (aged 16-24 years) strongly preferring full NHS funding for those under 18 years old (p = 0.007, 95% CI: 0.57-0.09) who dislike smiling in public, especially where self-esteem and confidence are impaired (p = 0.002, 95% CI: 0.16-0.71). Participants with high annual income had the highest preference for the NHS to fund treatment regardless of income (p = 0.02, 95% CI: 0.13-1.47) and placed an onus on addressing developmental anomalies (p = 0.004, 95% CI: 0.22-1.15). In total, 159 (63.6%) of those who would undergo treatment were willing to pay for it, with the majority (88%) open to paying up to £2,000 and only three participants stating the NHS should not contribute towards the cost of orthodontic treatment.Conclusions Based on this pilot study, key factors influencing the decision to undergo treatment included family income, cost, the aetiology of malocclusion, age and self-esteem/confidence. It was felt that free NHS-based treatment should be given priority where self-esteem and confidence are impaired among young people. Further research to inform the priorities underpinning the provision of dental care and orthodontic treatment within the NHS is required.

Objectives To determine the UK prevalence of behavioral problems in 5-year-old children born with isolated or syndromic cleft lip and/or palate (CL/P) compared to the general population and identify potentially associated factors. Design Observational study using questionnaire data from the Cleft Collective 5-Year-Old Cohort study and three general population samples. Main Outcome Measure The Strengths and Difficulties Questionnaire (SDQ). Participants Mothers of children (age: 4.9-6.8 years) born with CL/P ( n = 325). UK general population cohorts for SDQ scores were: Millennium Cohort Study (MCS) ( n = 12 511), Office of National Statistics (ONS) normative school-age SDQ data ( n = 5855), and Avon Longitudinal Study of Parents and Children (ALSPAC) ( n = 9386). Results By maternal report, 14.2% of children born with CL/P were above clinical cut-off for behavioral problems, which was more likely than in general population samples: 7.5% of MCS (OR = 2.05 [1.49-2.82], P < 0.001), 9.8% of ONS (OR = 1.52 [1.10-2.09], P = 0.008), and 6.6% of ALSPAC (OR = 2.34 [1.70-3.24], P < 0.001). Children in the Cleft Collective had higher odds for hyperactivity, emotional and peer problems, and less prosocial behaviors. Maternal stress, lower maternal health-related quality of life and family functioning, receiving government income support, and maternal smoking showed evidence of association (OR range: 4.41-10.13) with behavioral problems, along with maternal relationship status, younger age, and lower education (OR range: 2.34-3.73). Conclusions Findings suggest elevated levels of behavioral problems in children born with CL/P compared to the general population with several associated maternal factors similar to the general population.

Objective To assess whether women who experience stressful life events during the periconceptional period are at higher risk of giving birth to a baby with an orofacial cleft (OFC). Design Systematic review and meta-analysis of studies reporting the proportion of babies born with OFC to mothers exposed and unexposed to population-level or personal-level stressful life events during the periconceptional period. Six electronic databases were searched from inception to August 2020. Risk of bias was assessed using the Newcastle-Ottawa scale. Odds ratios (ORs) for the odds of OFC in babies of exposed mothers relative to unexposed controls were extracted and/or calculated. Random effects meta-analysis was undertaken, stratified by cleft subtype. Results Of 12 eligible studies, 8 examined experience of personal events and 4 examined population-level events. Studies demonstrated low-moderate risk of bias and there was indication of publication bias. There was some evidence that personal stressful life events were associated with greater odds of cleft lip and/or palate (six studies, OR 1.63, 95% confidence interval (CI) 1.16, 2.30, P = 0.001) and cleft palate only (six studies, OR 1.45, 95% CI 1.02, 2.06, P = 0.04). Population-level events were associated with higher odds of OFC in studies that did not specify subtype (three studies, OR 1.64, 95% CI 1.19, 2.25, P = 0.002), but subtype stratified analyses were underpowered. Heterogeneity was high. Conclusions Limited evidence indicated a weak positive association between maternal stressful life events during the periconceptional period and risk of OFC in the offspring, but further studies with greater consistency in research design are needed.

Objectives To determine the priorities of patients and dental professionals concerning NHS dental treatments, the factors influencing prioritisation and the willingness to contribute towards the cost of NHS dental treatments. Methods Focus groups and interviews involving patients and practitioners informed the development of a piloted questionnaire concerning the priorities for NHS dental treatments. Patients attending three purposively selected dental settings in London and Kent, as well as dental professionals working within a large London dental hospital were recruited to participate in this initial qualitative phase. Qualitative interviews were audiotaped, transcribed verbatim and analysed using the framework approach. Subsequently, another sample of patients and dental professionals within the three dental settings and dental hospital completed a questionnaire. Regression models were used to determine the predictors of perceived priorities and willingness to contribute to NHS dental costs based on the questionnaire data. Results Three focus groups (n = 9) and one semi-structured interview with patients and one focus group of dental professionals (four general dental practitioners and two dental nurses) were conducted. Participants prioritised NHS dental treatments that improve quality of life and social wellbeing. Factors influencing the prioritisation of NHS dental treatments included: individual responsibility for oral health care; concerns about self-esteem and confidence; age-related issues; and the role of treatment in prevention of future dental and general health problems, with financial concerns underpinning these themes. Out of the 455 questionnaires completed, 414 (383 patients and 31 general dental practitioners) were included in the analysis. The provision of emergency dental treatment for children was afforded the highest priority among both patients (59%) and dentists (74.2%). Both groups of participants felt that full funding for most NHS dental treatments should be prioritised for children (<18 years old) rather than adults (p <0.05). Conclusion Participants prioritised NHS dental treatments that would improve social wellbeing and quality of life, with an emphasis on full coverage for NHS treatment for children and young people. Policy makers should account for these preferences in the planning of NHS dental services.

A systematic review and meta-analysis to determine the association between active maternal smoking and cleft lip and palate etiology. Medline, Embase, Web of Science and the Cochrane Library from inception to November, 2020. Observational studies of cigarette smoking habits in pregnant women. Outcomes included cleft lip and/or palate, cleft lip ± palate and cleft palate only. Publication bias analyses were performed and the Newcastle Ottawa scales were used to assess study quality. Fixed or random effect models were used in the meta-analysis, dependent on risk of statistical heterogeneity. Forty-five studies were eligible for inclusion of which 11 were cohort and 34 were case–control studies. Sixteen studies were of sufficient standard for inclusion in the meta-analysis. The summary odds ratio for the association between smoking and cleft lip and/or palate was 1.42 (95%CI 1.27-1.59) with a population attributable fraction of 4% (95%CI 3%-5%). There was limited evidence to show a dose–response effect of smoking. This review reports a moderate association between maternal smoking and orofacial cleft but the overall quality of the conventional observational studies included was poor. There is a need for high quality and novel research strategies to further define the role of smoking in the etiology of cleft lip and palate.

Background/Purpose: Covid-19 has impacted on the delivery of health care and education to children. Those with underlying medical conditions, including children born with clefts, are particularly vulnerable. During the lockdown period in the United Kingdom, some interventions were delivered using remote methods and other innovative ways. Nevertheless, delays in surgery and other treatments may have increased anxiety in parents and children and adversely influenced outcomes. Previous reports show that children born with clefts typically perform worse on measures of educational attainment relative to their peers. During the pandemic, home schooling replaced conventional teaching and it is not yet known what impact that has had specifically for children born with clefts. The primary aim of this study was to determine patients' and families' views of cleft care and educational support received over lockdown. The secondary aim was to identify how the well-being of children with cleft was affected during this period. Methods/Description: The cleft collective study is a large prospective longitudinal UK cohort study of children born with cleft lip and/or palate available as a resource to researchers. To date, the study has recruited over 9000 participants from over 3000 families. Children within the study are now aged between 0 and 15. The Cleft Collective recently invited parent participants to complete a questionnaire about the impact of Covid-19. A mixed methods approach was used to address the study aims. Free-text data from the questionnaire was used to undertake a thematic analysis on responses to questions about interventions received, delayed treatment, educational support, and more general questions about Covid-19 and lockdown. Data on the child's emotions and worries were collected retrospectively for the first (complete) and second (partial) phases of lockdown. The emotions and worries data were analyzed using a repeated measures technique for the 2 different time periods. Data were also compared as independent groups, to identify which characteristics helped children cope better during lockdown. Results: Over 1100 families responded and preliminary analysis has identified several themes from the data so far. These include both negative and positive experiences of participants during lockdown. The data also highlight the concerns of parents, especially with regard to delays in receiving face-to-face speech and language therapy. Analysis is ongoing and will be reported in full at the time of the presentation. Conclusions: In conclusion, this study provides real-time data to cleft clinicians and education providers highlighting parental concerns and interventions that have gone well. The results provide important information for cleft care in the event that further lockdowns are imposed. The study findings also identify which groups may need additional support with regard to a child's well-being and anxieties. Future analyses will determine the impact of the pandemic and associated lockdown on longer term outcomes.