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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.

Objective Speech sound disorder (SSD) describes a ‘persistent difficulty with speech sound production that interferes with speech intelligibility or prevents verbal communication’. There is a need to establish which care pathways are most effective and efficient for children with SSD. Comparison of care pathways requires clearly defined, evidence-based, interventions and agreement on how to measure the outcomes. At present, no definitive list of assessments, interventions or outcomes exists. The objective of this umbrella review paper is to provide a rigorous and detailed list of assessments, interventions and outcomes which target SSD in children.Design In December 2022, a systematic search of Ovid Medline, OVID Embase, CINAHL, PsycInfo and Cochrane and a number of grey literature platforms were undertaken. 18 reviews were included, and subsequently 415 primary research articles were assessed for data related to assessments, interventions or outcomes. The AMSTAR (Assessing the Methodological Quality of Systematic Reviews) framework was used to assess the quality of the retained reviews.Setting Reviews were retained which took place in any setting.Participants The population is children of any age with a diagnosis of SSD of unknown origin.Primary and secondary outcome measures Reviews reporting outcomes, assessment and interventions for children with SSD.Results Extraction and analysis identified 37 assessments, 46 interventions and 30 outcome measures used in research reporting of SSD. Not all of the listed outcomes were linked to specific outcome measurement tools, but these were measurable through the use of one or more of the assessments extracted from the retained reviews.Conclusions The findings of this review will be used to develop a Core Outcome Set for children with SSD. The findings are part of a rigorous process essential for advancing healthcare research and practice in the specific area of speech and language therapy for children with SSD.PROSPERO registration number CRD42022316284.

Background:. There is debate amongst surgeons regarding the use of antibiotics to prevent fistulae after palatoplasty. Prescribing should be evidence based, as antibiotic stewardship is integral to reducing antibiotic resistance. Our aim was to determine whether differing perioperative regimens affect the prevalence of postoperative fistulae. Methods:. The sample comprised participants from the Cleft Collective who had undergone palatoplasty. Participants were recruited across all 16 UK cleft centers between 2013 and 2021. The exposure was perioperative antibiotic regimen prescribed at the time of palatoplasty. The primary outcome was the presence of palatal fistula. Results:. Fistula data were available for 167 participants when exploring antibiotic regimen and for 159 when exploring antibiotic agent. There was no evidence to suggest a difference in fistula rate between those receiving antibiotics on induction only versus as an inpatient or up to 7 days postoperatively (χ2 = 4.57; P = 0.10). There was no evidence to suggest a difference in fistula rate between those who received co-amoxiclav and those who had an alternative antibiotic (χ2 = 0.16; P = 0.69). Postoperative fistulae increased with the extent of the cleft (χ2 = 20.39; P < 0.001). When adjusting for cleft type, no evidence of an association between antibiotic regimen and fistulae was found (inpatient antibiotics: OR 1.36; 95% confidence interval, 0.53–3.51; antibiotics up to 7 days postoperatively: OR 0.68; 95% confidence interval, 0.26–1.80). Conclusions:. The choice of antibiotic and dosing regimen does not influence the formation of postoperative fistulae. These results should be supported by interventional trials.

Abstract Background It is well-documented that children with Developmental Language Disorder (DLD) have a higher likelihood of experiencing anxiety, as well as other socio-emotional and behavioural (SEB) difficulties. Despite this, there is little consensus as to how these difficulties manifest. This study aims to understand the prevalence of broader SEB difficulties and anxiety, informing intervention development by understanding the relationships between them. Methods A mixed-methods, case–control study was conducted. First, an online survey was completed by 107 parents of either children with DLD (“DLD sample”; n = 57) or typically developing children (“typical sample”; n = 50), aged 6–12 years old. Binary SEB statements informed by previous qualitative work (e.g. “my child requires routine/sameness”; “my child has frequent tantrums”) provided an insight into the prevalence of SEB difficulties in both DLD and typical samples. Validated measures of anxiety, emotion regulation, intolerance of uncertainty, insistence on sameness, family stress and coping mechanisms were also collected. Correlation and mediation analyses were run using these validated measures to understand the manifestation of anxiety in children with DLD in more detail. Qualitative interviews were then carried out with a select panel of survey respondents (n = 4). Results The DLD sample scored significantly higher on all binary SEB statements than the typical sample: experiencing anxiety (80.7%, p

BackgroundIn order to leverage the potential benefits of technology to speech and language therapy language assessment processes, large samples of naturalistic language data must be collected and analysed. These samples enable the development and testing of novel software applications with data relevant to their intended clinical application. However, the collection and analysis of such data can be costly and time-consuming. This paper describes the development of a novel application designed to elicit and analyse young children’s story retell narratives to provide metrics regarding the child’s use of grammatical structures (micro-structure) and story grammar (macro-structure elements). Key aspects for development were (1) methods to collect story retells, ensure accurate transcription and segmentation of utterances; (2) testing the reliability of the application to analyse micro-structure elements in children’s story retells and (3) development of an algorithm to analyse narrative macro-structure elements.MethodsA co-design process was used to design an app which would be used to gather story retell samples from children using mobile technology. A citizen science approach using mainstream marketing via online channels, the media and billboard ads was used to encourage participation from children across the United Kingdom. A stratified sampling framework was used to ensure a representative sample was obtained across age, gender and five bands of socio-economic disadvantage using partial postcodes and the relevant indices of deprivation. Trained Research Associates (RA) completed transcription and micro and macro-structure analysis of the language samples. Methods to improve transcriptions produced by automated speech recognition were developed to enable reliable analysis. RA micro-structure analyses were compared to those generated by the digital application to test its reliability using intra-class correlation (ICC). RA macro-structure analyses were used to train an algorithm to produce macro-structure metrics. Finally, results from the macro-structure algorithm were compared against a subset of RA macro-structure analyses not used in training to test its reliability using ICC.ResultsA total of 4,517 profiles were made in the app used in data collection and from these participants a final set of 599 were drawn which fulfilled the stratified sampling criteria. The story retells ranged from 35.66 s to 251.4 s in length and had word counts ranging from 37 to 496, with a mean of 148.29 words. ICC between the RA and application micro-structure analyses ranged from 0.213 to 1.0 with 41 out of a total of 44 comparisons reaching ‘good’ (0.70–0.90) or ‘excellent’ (>0.90) levels of reliability. ICC between the RA and application macro-structure features were completed for 85 samples not used in training the algorithm. ICC ranged from 0.5577 to 0.939 with 5 out of 7 metrics being ‘good’ or better.ConclusionWork to date has demonstrated the potential of semi-automated transcription and linguistic analyses to provide reliable, detailed and informative narrative language analysis for young children and for the use of citizen science based approaches using mobile technologies to collect representative and informative research data. Clinical evaluation of this new app is ongoing, so we do not yet have data documenting its developmental or clinical sensitivity and specificity.

Abstract Purpose Social, emotional and behavioural difficulties (SEBD) in childhood are associated with negative consequences across the life course. Children with developmental language disorder have been identified as being at risk of developing SEBD but it is unclear whether a similar risk exists for children with speech sound disorder, a condition which impacts on children's ability to make themselves understood and has been shown to be associated with poor educational outcomes. Methods Participants were children who attended the 8‐year‐old clinic in the Avon Longitudinal Study of Parents and Children (N = 7390). Children with speech sound disorder that had persisted beyond the period of typical speech acquisition (persistent speech disorder [PSD]) at age 8 were identified from recordings and transcriptions of speech samples (N = 263). Parent‐, teacher‐ and child‐reported questionnaires and interviews including the Strengths and Difficulties Questionnaire, Short Moods and Feelings Questionnaire and measures for antisocial and risk‐taking behaviour were used to provide outcome scores for SEBD at 10–14 years in a series of regression analyses. Results Following adjustment for biological sex, socio‐economic status and Intelligence Quotient, children with PSD at age 8 were more likely to show peer problems at age 10–11 years compared with their peers, as reported by teachers and parents. Teachers were more likely to report problems with emotionality. Children with PSD were no more likely to report symptoms of depression than their peers. No associations were observed between PSD, risk of antisocial behaviour, trying alcohol at age 10 or smoking cigarettes at age 14. Conclusions Children with PSD may be at risk in terms of their peer relationships. This could impact on their wellbeing and, while not observed at this age, may lead to depressive symptoms in older childhood and adolescence. There is also the potential that these symptoms may impact on educational outcomes.

Introduction Speech sound disorder (SSD) describes a ‘persistent difficulty with speech sound production that interferes with speech intelligibility or prevents verbal communication’. There is a need to establish which care pathways are most effective and efficient for children with SSD. Comparison of care pathways requires clearly defined, evidence-based interventions and agreement on how to measure the outcomes. At present, no list of assessments, interventions or outcomes exists.The objective of this paper is to provide a rigorous and detailed protocol for an umbrella review of assessments, interventions and outcomes that target SSD in children. The protocol details the development of a search strategy and trial of an extraction tool.Methods and analyses The umbrella review has been registered with PROSPERO (CRD42022316284). Papers included can use a review methodology of any sort but must include children of any age, with an SSD of unknown origin. In accordance with the Joanna Briggs Institute scoping review methods guidelines, an initial search of the Ovid Emcare and Ovid Medline databases was conducted. Following this, a final search strategy for these databases were produced. A draft extraction form was developed.Ethics and dissemination Ethical approval is not needed for an umbrella review protocol. Following the systematic development of an initial search strategy and extraction form, an umbrella review of this topic can take place. Dissemination of findings will be through peer-reviewed publications, social media, and patient and public engagement.

Abstract Speech sound disorders (SSD) manifest as difficulties in phonological memory and awareness, oral motor function, language, vocabulary, reading, and spelling. Families enriched for SSD are rare, and typically display a cluster of deficits. We conducted a genome-wide association study (GWAS) in 435 children from 148 families in the Cleveland Family Speech and Reading study (CFSRS), examining 16 variables representing 6 domains. Replication was conducted using the Avon Longitudinal Study of Parents and Children (ALSPAC). We identified 18 significant loci (combined p

Abstract Background This systematic review aims to inform the development of a screening tool which pre-operatively predicts which children are likely to develop velopharyngeal insufficiency, one of the causes of poor speech outcomes, following cleft palate repair. This would be highly beneficial as it would inform pre-operative counselling of parents, allow targeted speech and language therapy, and enable meaningful comparison of outcomes between surgeons, techniques, and institutions. Currently, it is unclear which factors influence speech outcomes. A systematic review investigating the non-interventional factors which potentially influence speech outcomes following cleft palate repair is warranted. This may be illuminating in itself or provide foundations for future studies. Methods A systematic review will be carried out according to Cochrane methodology and reported according to PRISMA guidelines (PLoS Med 6: e1000097, 2009). Systematic review software will be used to facilitate three-stage screening by two independent reviewers experienced in cleft lip and palate. Thereafter, data extraction and GRADE assessment will be performed in duplicate by five independent reviewers experienced in cleft lip and palate. Studies reporting the proportion of patients who were recommended or underwent secondary speech surgery for velopharyngeal insufficiency following primary surgery for cleft palate will be included. The study findings will be tabulated and summarised. The primary outcome measure will be further speech surgery (either recommended or performed). The secondary outcome measure will be perceptual speech assessment for the presence of velopharyngeal insufficiency. A meta-analysis is planned. However, if this is not possible, due to the anticipated marked heterogeneity of study characteristics, pre-operative assessment, and the recorded outcome measures, a narrative synthesis will be undertaken. Discussion This systematic review may provide sufficient data to inform the development of a screening tool to predict the risk of velopharyngeal insufficiency prior to cleft palate repair. However, it is anticipated that these findings will provide the foundation for future studies in this area. Systematic review registration Registered on 19 December 2016 with PROSPERO CRD42017051624

BackgroundBoth active and passive cigarette smoking have previously been associated with orofacial cleft aetiology. We aimed to analyse the impact of declining active smoking prevalence and the implementation of smoke-free legislation on the incidence of children born with a cleft lip and/or palate within the United Kingdom.Methods and findingsWe conducted regression analysis using national administrative data in the United Kingdom between 2000-2018. The main outcome measure was orofacial cleft incidence, reported annually for England, Wales and Northern Ireland and separately for Scotland. First, we conducted an ecological study with longitudinal time-series analysis using smoking prevalence data for females over 16 years of age. Second, we used a natural experiment design with interrupted time-series analysis to assess the impact of smoke-free legislation. Over the study period, the annual incidence of orofacial cleft per 10,000 live births ranged from 14.2-16.2 in England, Wales and Northern Ireland and 13.4-18.8 in Scotland. The proportion of active smokers amongst females in the United Kingdom declined by 37% during the study period. Adjusted regression analysis did not show a correlation between the proportion of active smokers and orofacial cleft incidence in either dataset, although we were unable to exclude a modest effect of the magnitude seen in individual-level observational studies. The data in England, Wales and Northern Ireland suggested an 8% reduction in orofacial cleft incidence (RR 0.92, 95%CI 0.85 to 0.99; P = 0.024) following the implementation of smoke-free legislation. In Scotland, there was weak evidence for an increase in orofacial cleft incidence following smoke-free legislation (RR 1.16, 95%CI 0.94 to 1.44; P = 0.173).ConclusionsThese two ecological studies offer a novel insight into the influence of smoking in orofacial cleft aetiology, adding to the evidence base from individual-level studies. Our results suggest that smoke-free legislation may have reduced orofacial cleft incidence in England, Wales and Northern Ireland.

Introduction The Cleft Collective Cohort study is the world's largest multidisciplinary cleft lip/palate research programme. Despite being one of the most common birth anomalies, the causes of clefting are unknown. Treatment involves a considerable burden of care from birth onwards, together with a variety of social and psychological challenges. Objectives and Approach Our aim is to create the infrastructure and resources necessary to gain important new knowledge that will advance our understanding of the causes of cleft lip/palate, inform treatment and improve the lives of people born with cleft; data linkage is a key aspect of this. There are challenges associated with linking to multiple data sources (NHS Digital, Cleft National Registry). However, using the consent obtained, we are also able to link directly to the participant’s medical notes held by their NHS cleft team; enabling us to access a variety of data, including free text. Results The study already collects social and demographic data via questionnaires and genetic data from biological samples. Data linkage enriches this but also enables us to validate and address missing data problems. However, linkage to external sources brings many challenges, including, governance, costs, access issues. By gaining direct access to cleft team medical notes these issues are significantly reduced and provide us with rich phenotype data that cannot be obtained elsewhere, for example, via ‘read codes’ within electronic medical records. By tailoring our own data collection tool we can collect specific cleft data to enhance the resource and allow for subtype analyses. This process can be repeated throughout the duration of this longitudinal study for subsequent data. Conclusion/Implications Data linkage is a valuable resource but comes with many challenges. One route to overcome many of these issues is by accessing free text data directly from participants medical notes. The richness of these data allows for more in depth phenotypic analyses.