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Background: Adolescent self-harm is a major public health issue internationally. Various factors associated with adolescent self-harm have been identified, including being bullied and experiencing mental health problems. Stuttering and speech sound disorder are associated with both of these factors. It was hypothesized that both stuttering and speech sound disorder would be associated with self-harm. This is the first study to explore the relationship between communication disorders and adolescent self-harm. Method: Secondary analysis of a large, longitudinal, prospective, community sample, the Avon Longitudinal Study of Parents and Children, was carried out. Clinicians identified children who stuttered or exhibited speech sound disorder at the age of 8 years. When the cohort members were 16 years old, they were asked to complete a questionnaire about self-harm. Multinomial logistic regression was used to examine the associations between stuttering and speech sound disorder and the self-harm outcomes, adjusting for other relevant factors. Results: Of 3,824 participants with data for both speech status and self-harm, 94 (2.5%; 95% confidence interval [CI; 2.0, 3.0]) stuttered at 8 years of age and 127 (3.3%; 95% CI [2.8, 3.9]) displayed speech sound disorder. Speech sound disorder at the age of 8 years was associated with self-harm with suicidal intent in both unadjusted and adjusted models. Differences between the adjusted and unadjusted models were small, suggesting that speech sound disorder is largely an independent risk factor for self-harm with suicidal intent. Stuttering at the age of 8 years was not associated with adolescent self-harm, and there was no association between speech sound disorder and self-harm without suicidal intent. Conclusion: Compared with individuals without speech sound disorder, adolescents with speech sound disorder at the age of 8 years have twice the risk of reporting self-harm with suicidal intent, even when other important predictors are taken into account. Supplemental Material: https://doi.org/10.23641/asha.22573030

Purpose: This study aimed to achieve a consensus on a diagnostic protocol, classification system, and subtype definitions for the differential diagnosis of speech sound disorder of unknown origin in the United Kingdom. Method: A mixed methods participatory design was used. Five services from the UK provided all paperwork, including guidelines and care pathways, related to Speech Sound Disorder for content analysis. Two participatory workshops with six speech and language therapists from these five services were used to discuss and agree: 1. A classification system, 2. Subtype labels and definitions, and 3. A diagnostic protocol for speech sound disorder suitable for use in the UK context. Result: Participants agreed that the Differential Diagnosis System (Dodd, 2014) was suitable for use in the UK. This system comprises five speech sound disorder subtypes. Participants suggested minor changes to the definitions of these subtypes to make them more suitable for implementation in a clinical context. A minimum diagnostic protocol, with additional assessment for children with more complex or severe speech sound disorder, was agreed. Conclusion: A consensus diagnostic protocol, classification system, and subtype names and definitions was reached and is broadly in line with Dodd (2014). Future work will implement this in the national health service in the UK

Purpose: The purpose of this study was to examine parent/caregiver perspectives and experiences of speech-language pathology provision during the COVID-19 pandemic for children born with cleft palate. Method: An online questionnaire to parents of children born with cleft palate asked about delays and changes to speech-language pathology provision during the first U.K. national lockdown. Parents were also asked their views on the effectiveness of online speech-language pathology provision. Analysis considered variation in speech-language pathology provision by region. Chi-square and Mann–Whitney U tests examined associations between speech-language pathology provision and socioeconomic status and child age. Free text responses were analyzed using qualitative content analysis. Results: Three hundred fifty-six (39.3%) children were receiving speech-language pathology intervention before the first national lockdown. A further 49 (9.0%) were due to start speech-language pathology intervention during the lockdown. Speech-language pathology provision varied both nationally and within smaller geographical regions. Overall, 146 (42.6%) children continued to receive speech-language pathology and 197 (57.4%) had intervention delayed. There was no association between delayed speech-language pathology and socioeconomic status. Older children were more likely to experience delayed speech-language pathology provision ( p = .004). Qualitative analysis revealed concerns about access to speech-language pathology, challenges with adequate devices to access online provision, technological problems, and child engagement in online provision. Parents reported online provision as being “better than nothing.” Conclusions: Parents/caregivers reported delays to speech-language pathology provision during the first lockdown, but this varied geographically and was more prevalent for older children. Concerns about access to speech-language pathology provision were raised, including challenges regarding online provision. Follow-on work will consider the impact of the delays experienced on longer term outcomes.

Background It is well-documented that children with Developmental Language Disorder (DLD) experience socio-emotional difficulties. Despite this, there is little consensus as to how these difficulties manifest. This study aims to understand their prevalence, and inform 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. The online survey comprised of standardised psychosocial measures, binary psychosocial statements (generated from previous qualitative work) and both family stress and coping mechanisms. Two separate mediation models were run to understand the underlying mechanisms behind (1) anxiety, and (2) social frustration; separate correlation analyses were used to determine the relationships between other psychosocial and familial variables. Qualitative interviews were then carried out with a small subset of the survey respondents (n = 4). Results The DLD sample scored significantly higher on all psychosocial statements than the typical sample. Experiencing anxiety (80.7%, p

BackgroundThe publication of phase 2 of the CATALISE project in 2017 clarified terminology for children with developmental language disorder (DLD) or delay but unintentionally muddied the water for children with unintelligible speech. A diagnostic label of DLD (phonology) indicates poor prognosis and phonological disorder that persists into middle childhood. However, in contrast to other diagnostic labels that fall under the overarching term of speech sound disorder (SSD), DLD (phonology) does not elucidate the characteristics of the child’s speech nor does it point us in the direction of appropriate intervention. AimsThe aim of this paper is to discuss terminology in SSD leading to an evidence based model which builds on the model of DLD developed in CATALISE, supports descriptive diagnosis and signposts intervention. MethodsFollowing a focused review of literature proposing or describing terminology for SSD, an expert group of researchers in developmental SSD proposed a revised model of existing terminology. Groups of UK speech and language therapists (SLTs) who provide services for children with SSD were asked to comment on its acceptability and feasibility. Discussion A three level terminology model was developed. This comprised an overarching Level 1 term; Level 2 terms that differentiated SSD of unknown origin from SSD with associated or underlying conditions; and specific diagnostic terms at Level 3 to support further assessment and intervention decisions. Consulted SLTs generally expressed agreement with the proposed terminology and a willingness to adopt it in practice.ConclusionsExisting terminology for childhood SSD provides a good basis for clinical decision-making. A modified version of Dodd’s (2005) terminology was found to be acceptable to UK SLTs. There is an evident overlap of SSD with CATALISE terminology. However more detailed and specialist terminology than “DLD (phonology)” is required to support clinical decision-making. It is proposed that endorsement by the UK Royal College of Speech and Language Therapists (RCSLT) would obviate the need for a Delphi process.

Objective: To investigate the early communication behaviors in infants with nonsyndromic isolated cleft palate (iCP) and Robin sequence (RS). Design: Group comparison using parent report. Participants: There were 106 participants included in this study. Two groups were selected from the UK Cleft Collective resource. Parents had completed the Language ENvironment Analysis Developmental Snapshot questionnaire when their child turned 13 months. There were 78 participants in the iCP group and 28 in the RS group. Main Outcome Measure(s): Total number of communication behaviors reported on the questionnaire. Subdomains for expressive and receptive language and social communication behaviors were also analyzed. Results: There were no statistically significant group differences. Parents of infants with RS reported fewer later communication behaviors compared to the iCP group. Infants in both groups had fewer communication behaviors compared to the normative sample. Across the whole sample, post hoc analysis revealed a significant correlation between severity of the cleft and social communication behaviors and expressive but not receptive language. Infants with a cleft of the hard and soft palate were more likely to be in the RS group (odds ratio: 7.04 [95% CI: 1.55-32.04]; P = .01). Conclusions: Both groups reported similar levels of early communication. Some divergence of more complex language skills was seen, although there were no significant group differences. A relationship with the diagnosis of a cleft of the hard or soft palate with expressive language behaviors was found. Further study into the impact of cleft severity on early speech development and the relationship with later language skills is needed along with longitudinal follow-up of this population.

ObjectivesTo determine the prevalence of syndromic Robin sequence (RS) in the UK and if this group of patients had an increased need for airway and feeding management compared with a non-syndromic RS cohort.DesignA prospective national multicentre study of cases submitted to the Cleft Collective cohort studies.SettingSpecialist cleft services in the UK.Patients259 participants who fulfilled the diagnosis of RS. This group was compared with 548 participants with cleft palate only (CPO).Main outcome measuresThe primary outcome measure was the presence of a syndrome in patients with RS and CPO. Secondary outcome measures included the use of airway and feeding adjuncts.ResultsAn associated syndrome was seen in 28% of patients with RS and 14% of patients with CPO. The most common syndrome for the RS group was Stickler syndrome (27%). Syndromic status was significantly higher among patients with RS compared with those with CPO (OR 2.36, 95% CI 1.65 to 3.39; pConclusionThe presence of a syndrome has implications for management of patients with RS. Early identification of a syndrome may help prevent the consequences of a missed syndromic diagnosis. Routine ophthalmological and genetic screening for Stickler syndrome should be mandatory for all patients with RS.

Background Previous studies have found that children born with a non-syndromic orofacial cleft have lower-than-average educational attainment. Differences could be due to a genetic predisposition to low intelligence and academic performance, factors arising due to the cleft phenotype (such as social stigmatization, impaired speech/language development) or confounding by the prenatal environment. A clearer understanding of this mechanism will inform interventions to improve educational attainment in individuals born with a cleft, which could substantially improve their quality of life. We assessed evidence for the hypothesis that common variant genetic liability to non-syndromic cleft lip with or without cleft palate (nsCL/P) influences educational attainment. Methods We performed a genome-wide association study (GWAS) meta-analysis of nsCL/P with 1692 nsCL/P cases and 4259 parental and unrelated controls. Using GWAS summary statistics, we performed Linkage Disequilibrium (LD)-score regression to estimate the genetic correlation between nsCL/P, educational attainment (GWAS n = 766 345) and intelligence (GWAS n = 257 828). We used two-sample Mendelian randomization to evaluate the causal effects of genetic liability to nsCL/P on educational attainment and intelligence. Results There was limited evidence for shared genetic aetiology or causal relationships between nsCL/P and educational attainment [genetic correlation (rg) −0.05, 95% confidence interval (CI) −0.12 to 0.01, P 0.13; MR estimate (βMR) −0.002, 95% CI −0.009 to 0.006, P 0.679) or intelligence (rg −0.04, 95% CI −0.13 to 0.04, P 0.34; βMR −0.009, 95% CI −0.02 to 0.002, P 0.11). Conclusions Common variants are unlikely to predispose individuals born with nsCL/P to low educational attainment or intelligence. This is an important first step towards understanding the aetiology of low educational attainment in this group.

Background: Children with and without speech sound disorders (SSDs) are exposed to different patterns of infant feeding (breast/bottle-feeding) and may or may not engage in non-nutritive sucking (NNS) (pacifier/digit-sucking). Sucking and speech use similar oral musculature and structures, therefore it is possible that early sucking patterns may impact early speech sound development. The objective of this review is to synthesise the current evidence on the influence of feeding and NNS on the speech sound development of healthy full-term children. Summary: Electronic databases (PubMed, NHS CRD, EMBASE, MEDLINE) were searched using terms specific to feeding, NNS and speech sound development. All methodologies were considered. Studies were assessed for inclusion and quality by 2 reviewers. Of 1,031 initial results, 751 records were screened, and 5 primary studies were assessed for eligibility, 4 of which were included in the review. Evidence from the available literature on the relationship between feeding, NNS and speech sound development was inconsistent and inconclusive. An association between NNS duration and SSDs was the most consistent finding, reported by 3 of the 4 studies. Quality appraisal was carried out using the Appraisal Tool for Cross-Sectional Studies (AXIS). The included studies were found to be of moderate quality. Key Messages: This review found there is currently limited evidence on the relationship between feeding, NNS and speech sound development. Exploring this unclear relationship is important because of the overlapping physical mechanisms for feeding, NNS and speech production, and therefore the possibility that feeding and/or sucking behaviours may have the potential to impact on speech sound development. Further high-quality research into specific types of SSD using coherent clinically relevant assessment measures is needed to clarify the nature of the association between feeding, NNS and speech sound development, in order to inform and support families and health care professionals.

Purpose: The purpose of this study was to describe and examine parent views of speech-language pathology (SLP) for children born with cleft palate delivered via telemedicine during the COVID-19 pandemic in the United Kingdom (UK). Method: Parents were asked whether they found this method of delivery “very effective,” “somewhat effective,” or “not at all effective.” Free text was then invited. There were 212 responses. Ordinal chi-square, Kruskal–Wallis, or Fisher's exact tests examined associations between parent views of effectiveness and biological variables and socioeconomic status. Free text responses were analyzed using qualitative content analysis. Results: One hundred and forty (66.0%) respondents reported that SLP delivered via telemedicine was “somewhat effective,” 56 (26.4%) “very effective,” and 16 (7.6%) “not at all effective.” There was no evidence of an association between parent reported effectiveness and any of the explanatory variables. Parent-reported challenges impacting on effectiveness included technology issues and keeping their children engaged with sessions. Importantly, telemedicine was viewed as “better than nothing.” Conclusions: Most parents reported that they felt SLP delivered via telemedicine during the first few months of the COVID-19 pandemic in the UK was at least “somewhat effective.” It is important to interpret this in the context of there being no other method of service delivery during this time and that this study only represents families who were able to access SLP delivered via telemedicine. Further work is needed to identify which children with cleft palate might benefit from SLP delivered via telemedicine to inform postpandemic service provision.

IntroductionSpeech 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 analysesThe 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 disseminationEthical 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.

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.

Background Children with cleft palate with or without cleft lip (CP±L) are at high risk of problems with early speech sound production, and these difficulties can persist into later childhood. Early intervention could help to reduce the number of children whose problems become persistent. However, much research in the field to date has focused on older children. There is a need to determine if providing intervention during the phase of early typical speech development leads to better outcomes. Aims To review the evidence for the effectiveness of interventions targeting speech, delivered in the first 3 years of life for children with CP±L, and discuss factors such as intervention type, facilitator, dosage, outcome measures and the age of the child. Methods & procedures The systematic review was registered with PROSPERO (CRD42019121964). Eight bibliographic databases including CINAHL and MEDLINE were searched in August 2018. Studies were included if participants received speech and language interventions before 37 months and if they reported outcomes for speech. Two reviewers independently completed inclusion reviews, quality analysis and data extraction. Outcome & results The review included seven papers: one pilot randomized controlled trial, one controlled trial, four cohort studies and one case series report. Interventions largely took a naturalistic approach, namely focused stimulation and milieu teaching. The findings provide preliminary support for naturalistic interventions and suggest that these interventions can be delivered by parents with suitable training. Studies included in the review provided low-strength evidence with variation in both the type of intervention, the manner of delivery and in the risk of bias in the designs used. Conclusions & implications The papers included in this review suggest that early naturalistic interventions can have positive impacts on the speech development of children with CP±L. However, the reported methodological quality of the publications overall was weak, and the current evidence lacks clarity and specificity in terms of therapy technique, delivery and optimum age of delivery. Future research should use more robust methodological designs to determine whether early speech interventions are beneficial for children born with CP±L. What this paper adds What is already known on the subject Children with CP±L show difficulties with early speech development and often have restricted speech sound inventories. They may reach the canonical babbling stage later than children without CP±L and studies have shown that 20% of children with CP±L have speech which is considered unintelligible or barely intelligible at age 5. It has been proposed that early intervention can lessen the impact of CP±L on speech development. However, currently, the evidence for early interventions for children with CP±L is limited, with the majority of studies focusing on children aged 3 years and older. What this paper adds to existing knowledge This paper reviews the evidence for different types of early interventions for speech provided to children born with CP±L and whether these interventions are effective in supporting speech sound development. In this review, early intervention is defined as intervention provided to children in the first 3 years of life. This review describes intervention approaches and how they are delivered for this population. What are the potential or actual clinical implications of this work? In the UK, children born with CP±L and their families are supported by National Health Service (NHS) services over a 20-year period and speech and language therapy sessions may take place over many weeks and months. If providing early intervention in the first 3 years of life is effective, there is the potential for improved speech outcomes in early childhood and a reduced burden of care on children, families and services. This review considers the evidence for early speech intervention for children with CP±L in the first 3 years of life and identifies areas for future research.

Linguistic interactions between parents and their children are frequently studied to investigate how children acquire language. From observations, researchers have identified interaction strategies that foster children’s language development. In turn, interventions to support children’s early language skills employ styles of interaction derived from these observations. However, researchers have not often considered how the activity context selected forobservation may affect the language used, or whether these contexts reflect children’s diverse experiences.The aim of this scoping review was to explore the breadth of literature about language use across a range of activities. Included studies described linguistic outputs of parents and typically developing children (aged 1;0 to 5;11 years) and activity context(s). Searches were conducted in PsycInfo, Medline, CINAHL, ERIC-ProQuest and Google Scholar. From 16,718 records, 59 studies were retained. Studies were charted according to the population included, linguistic outputs recorded, activity contexts studied and the methodological design. To allow for comparison of results across activity contexts, five thematic categories were identified: play activities, book reading, naturalistic routines, media and methodological implications. Challenges for future research are discussed, including ways to ensure the ecological validity of findings by coupling naturalistic language recordings with data collected during diverse everyday activity contexts.

Objective This study describes primary surgical reconstructions performed for children born with a cleft lip and/or palate (CL ± P) in the United Kingdom (UK). Design Data forms completed at the time of surgery included details on timing, technique, and adjuncts used during the operative period. Demographic data on participants were validated via parental questionnaires. Setting Data were obtained from the Cleft Collective, a national longitudinal cohort study. Patients Between 2015 and 2021, 1782 Cleft Collective surgical forms were included, relating to the primary reconstructions of 1514 individual children. Results The median age at primary cheiloplasty was 4.3 months. Unilateral cleft lips (UCL) were reconstructed with an anatomical subunit approximation technique in 53%, whereas bilateral cleft lips (BCL) were reconstructed with a broader range of eponymous techniques. Clefts of the soft palate were reconstructed at a median age of 10.3 months with an intravelar veloplasty in 94% cases. Clefts of the hard palate were reconstructed with a vomer flap in 84% cases in a bimodal age distribution, relating to reconstruction carried out simultaneously with either lip or soft palate reconstruction. Antibiotics were used in 96% of cases, with an at-induction-only regimen used more commonly for cheiloplasties ( P Conclusions This study contributes to our understanding of current cleft surgical pathways in the UK and will provide a baseline for analysis of the effectiveness of utilized protocols.

ObjectivesA systematic review and meta-analysis to determine the association between active maternal smoking and cleft lip and palate etiology.Data SourcesMedline, Embase, Web of Science and the Cochrane database from inception to November 2020.Study selectionObservational studies of cigarette smoking habits in pregnant women. Outcomes included cleft lip and/or palate, cleft lip ± palate and cleft palate only.Data analysisPublication 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.ResultsForty-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 to 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.ConclusionsThis 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.

ObjectivesWe determined the prevalence of behavioural problems in 5-year-old children born with Cleft Lip and/or Palate (CL/P) and compared it to the prevalence in general population samples. We also identified risk factors for behavioural problems in children with CL/P.DesignObservational study using questionnaire data from the Cleft Collective (CC) 5-Year-Old cohort study and three general population samples.Main Outcome MeasureThe Strengths and Difficulties Questionnaire (SDQ) was used to measure behavioural problems.ParticipantsA total of 340 children born with CL/P whose mothers had completed the SDQ when their child was 5 years old. Published estimates from three large cohorts were used to approximate general population SDQ scores in the UK and these were used as comparison groups; Millennium Cohort Study (MCS) (n=12,511), Office of National Statistics (ONS) normative school-age SDQ data (n=5,855) and the Avon Longitudinal Study of Parents and Children (ALSPAC) (n=9,386).ResultsAn estimated 14.3% of 5-year-old children born with CL/P experienced behavioural problems. There was strong evidence to suggest children with CL/P were more likely to experience difficulties than children in the general population, as measured by SDQ total difficulties scores from all three population cohorts: MCS (OR = 2.07 [95% CI = 1.50-2.85]; PConclusionsOur findings suggest elevated levels of behavioural problems in children born with CL/P, particularly emotional difficulties in boys, compared to the general population and indicate several factors associated observationally with these difficulties.

Background: A number of studies have suggested that practitioners hold speech and language therapy (SLT) practice as tacit and consequently it is difficult for the therapist to describe. The current study uses a range of knowledge elicitation (KE) approaches, a technique not used before in SLT, as a way of accessing this tacit knowledge. There is currently no agreed framework that establishes key factors underpinning practice for preschool children with speech and language disorders. This paper attempts to address that gap. Aims: The aim of this study was to develop a framework of speech and language therapists’ practice when working with preschool children with developmental speech and language disorders (DS&LD). Methods and Procedures: A mixed methods approach was adopted for this study. Data were collected iteratively, from 245 speech and language therapists with experience of working with preschool children with DS&LD across sites in England, by means of focus groups and national events. There were three stages of data collection; local sites, specific interest groups and two national events. KE techniques were used to gather data, with initial data being collected in local site focus groups. Findings from groups were taken to subsequent larger groups where a combination of concept mapping, teach-back and sorting exercises, generated a more detailed description of practice, using discussion of consensus and disagreement to stimulate further exploration and definition and provide validatory evidence. Outcomes and Results: This paper provides a high-level framework of therapy for preschool children with DS&LD that makes practice explicit in this area. The framework proposes that therapists’ aims for this group of children fall into three categories: addressing children’s areas of impairment and skills; achieving functionally meaningful skills and carryover; supporting adults to provide a supportive communication environment. The exact configuration is shaped by the child’s context and needs. Conclusions and Implications: The framework highlights themes that are well researched in the literature (e.g. speech) and others that have been little studied (e.g. adult understanding), indicating a disconnect between research evidence and practice. The research also highlights the complex nature of interventions for preschool children with DS&LD and the importance therapists attribute to tailoring therapy to individual needs. The framework provides a scaffold for speech and language therapists to focus their clinical practice and encourages us as a profession to better understand and explore the gaps between research evidence and clinical practice, for preschool children with DS&LD.

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 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.

ObjectivesTo analyse the association between active and passive cigarette smoking and the incidence of children born with a cleft lip and/or palate within the United Kingdom.DesignFirst, a longitudinal time-series study was conducted with routinely collected smoking prevalence data for females over 16 years of age. Second, an interrupted time-series design was used as a natural experiment to assess the impact of smoke-free legislation using segmented Poisson regression.SettingUnited Kingdom.PopulationAll children born between 2000 and 2018 and their mothers.Main outcome measuresOrofacial cleft incidence, reported annually for England, Wales and Northern Ireland and separately for Scotland.ResultsOver 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 (95%CI 1% to 14%; P=0.026) following the implementation of smoke-free legislation. In Scotland, there was weak evidence for an increase in orofacial cleft incidence following smoke-free legislation.ConclusionThese 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.SUMMARY BOXSection 1: What is already known on this topicAn association between maternal active and passive smoking and orofacial cleft has been previously reported in conventional individual-level observational studiesEcological and natural experiment time-series designs have been successfully used to demonstrate the association between population-level exposures and other non-cleft health outcomesSection 2: What this study addsEcological studies within a single country with complete cleft registration data are unlikely to be sufficiently powered to detect a relationship between time-series population level trends in active smoking and the number of children born with orofacial cleft each year.In England, Wales and Northern Ireland our natural experiment study showed a reduction in the proportion of children born with an orofacial cleft following the implementation of smoke-free legislation, but this was not replicated in Scotland.This study exploits the systematic collection of population data in the UK and adds to our understanding of the role of smoking in orofacial clefting as well as demonstrating the impact of public health interventions.

BACKGROUND: Speech development requires intact and adequately functioning oral anatomy and cognitive 'speech processing' skills. There is evidence that speech input processing skills are associated with speech output problems in children not born with a cleft. Children born with cleft palate ± lip (CP±L) are at high risk of developing disordered speech output. Less is known about their speech input processing skills and whether they are associated with cleft-related speech sound disorder (SSD). AIMS: (1) To collate and evaluate studies reporting evidence regarding the speech input processing skills of children born with cleft palate in comparison with data from typically developing children or other comparison groups; and (2) to identify any available evidence regarding relationships between speech input processing skills and speech output in children born with CP±L. METHODS & PROCEDURES: Potentially relevant studies published up to November 2019 were identified from the following databases: Medline via Ovid, Embase via Ovid, Cinahl via Ebscohost, PsycInfo via Ebscohost, BNI via ProQuest, AMED via Ovid, Cochrane Library and Scopus. Inclusion criteria were: peer-reviewed articles published in scientific journals, any design, published in English, participants born with a CP±L aged up to age 18 years who completed speech input processing assessments compared with normative data and/or a control or other comparison group. Critical Appraisal Skills Programme (CASP) checklists were used to quality appraise included studies. OUTCOMES & RESULTS: Six studies were retained in the final review. There is some evidence that children born with CP±L perform less well than non-cleft controls on some speech input processing tasks and that specific input processing skills may be related to errors in the children's speech. Heterogeneity in relation to study groups and assessments used, as well as small sample sizes, limits generalization of findings. CONCLUSION & IMPLICATIONS: There is limited evidence regarding the speech input processing skills of children born with CP±L. There are indications that children born with CP+/L may have difficulty in some aspects of speech input processing in comparison with children not born with a cleft, and that difficulties with some speech input processing tasks may be specific to errors in children's speech output. Further research is required to develop our understanding of these skills in this population and any associations with speech output. WHAT THIS PAPER ADDS: What is already known on the subject Few studies have been published that examine aspects of speech input processing in children born with CP±L. Theoretical models of speech processing, and published studies, propose that speech input processing skills are associated with SSD in children who were not born with a cleft. However, it is less clear whether there is any association between speech input processing and cleft-related SSD. What this paper adds to existing knowledge This review systematically collates and evaluates the published, peer-reviewed evidence regarding speech input processing skills in children born with CP±L. The collated evidence indicates that some speech input processing skills differ between children with and without CP±L. There is some evidence, from a single study, that speech input processing of specific cleft speech characteristics (CSCs) may be associated with the presence of these CSCs in the speech output of some children born with CP±L. What are the potential or actual clinical implications of this work? While the evidence is currently limited, increasing our knowledge of speech input processing skills in children born with CP±L contributes to our clinical understanding of the nature of cleft-related SSD. The current evidence suggests that speech and language therapists should consider speech input processing skills when assessing children with cleft-related SSD to support intervention planning. Considering these skills in relation to literacy development in these children may also be important.

Objective: The mainstay of palatal repair in the United Kingdom is the intravelar veloplasty (IVVP). It is not always possible to align the oral mucosa in the midline to achieve tension-free repair. The addition of lateral relieving incisions may aid transposition of the oral mucosa to allow closure. The aim of this study was to explore cleft features that may predispose to a requirement for relieving incisions in order to allow palate closure. Design: We performed a national multiinstitutional retrospective study using data from the UK Cleft Collective cohort study. Patients: The study sample consisted of 474 patients who had undergone IVVP at the time of palatal closure across all 16 of the UK cleft units. Results: We found strong evidence for the requirement for relieving incisions in patients with an increased degree of clefting per the Veau classification ( P < .001), increasing palatal soft-edge width ( P < .001) and moderate evidence of an associated use in patients with Pierre Robin sequence ( P = .015). Insufficient data were available to explore the relationship between intertuberosity distance and the presence of fistula formation with the use of relieving incisions. Conclusions: The results of this study identify cleft features that increase the likelihood for requiring lateral relieving incisions to allow palatal closure. The degree to which the addition of relieving incisions to IVVP affects maxillary growth and speech outcomes is unknown. Further study is required to answer this important question.

BACKGROUND NHS case note data are a potential source of practice-based evidence which could be used to investigate the effectiveness of different interventions for individuals with a range of speech, language and communication needs. Consistency in pre- and post-intervention data as well as the collection of relevant variables would need to be demonstrated as a precursor to adopting this approach in future investigations of speech and language therapy intervention. AIMS To explore whether routine clinical data collection for children with speech sound disorder (SSD) could be a potential source for examining the effectiveness of intervention(s). METHODS & PROCEDURES We examined case notes from three UK NHS services, reviewing 174 sets of case notes and 234 blocks of therapy provided for school-age children with SSD. MAIN CONTRIBUTION We found there was significant variation in pre- and post-intervention data and variables collected by the services. The assessment data available in the case notes across all sites were insufficient to be used to compare the effectiveness of different interventions. Specific issues included lack of consistent reporting of pre- and post-intervention data, and use of a variety of both formal and informal assessment tools. CONCLUSIONS & IMPLICATIONS The case notes reviewed were from three sites and may not represent wider clinical practice, nevertheless the findings suggest the sample explored indicates the need for more consistent and contemporaneous collection of data for children with SSD to facilitate the investigation of different interventions in practice. Researchers should work with the clinical community to determine a minimal dataset that includes a core outcome set and potential variables. This should be feasible to collect in clinical practice and provide a dataset for future investigations of clinically relevant research questions. This would provide an invaluable resource to the clinical academic and research communities enabling research questions to be addressed that have the potential to lead to improved outcomes and more cost-effective services. WHAT THIS PAPER ADDS What is already known on the subject While there is some evidence for the efficacy of therapy for children with SSD, studies typically focus on very specific populations who meet strict selection criteria and take place in university clinics or laboratory-style settings which do not reflect typical clinical practice in the UK and elsewhere. An alternative approach to investigating the effectiveness of interventions would be to use NHS case note data. It is not clear from the existing literature whether case note data are sufficiently robust to facilitate such an analysis. What this paper adds to existing knowledge This study found that case note data, in particular assessment data, were highly variable across services and would be insufficient to compare different interventions for this population. Agreement on what should be included in a minimal dataset for children with SSD is required to maximize the potential for NHS clinical case notes to become a resource for future research. What are the actual or potential clinical implications of this work? This study indicates that current clinical practice in SLT for children with SSD is inconsistent with regards to the reporting of pre- and post-intervention assessment data and other important variables in case notes. We make the case for agreeing a minimal dataset with a need for clinicians to work with researchers to determine core outcomes and additional relevant data, which can be feasibly collected in clinical practice.

Objective:This study evaluated association between functional outcomes in children born with unilateral cleft lip and palate (UCLP) and educational attainment.Design:Cleft Care UK (CCUK) was a United Kingdom (UK) wide cross-sectional study.Setting:UK Cleft Teams (data collected from all UK sites providing centralized cleft services).Patients, Participants:Five-year olds born with nonsyndromic UCLP (n = 268).Main Outcome Measure(s):National tests for educational attainment Key Stage 1 (KS1) undertaken by children at age 7 were linked to CCUK data to describe differences in educational attainment. Associations between functional outcomes and KS1 results were evaluated using regression analysis. We adjusted for birth month, gender, and an area-based measure of socioeconomic status.Results:Data were available for 205 children with UCLP. These children scored lower than national average (NA) scores across all subject areas, with a 0.62 lower score observed in the Average Point Score (APS; P = .01). There was association between being in a lower category for a cleft related outcomes and poorer KS1 results, with a trend for poorer attainment with higher numbers of poor functional outcomes. Those with 3 or more poor outcomes had a −2.26 (−3.55 to −0.97) lower APS compared to those with 0 to 1 poor outcomes.Conclusions:Children born with UCLP have poorer educational attainment at age 7 across all subject areas though differences were modest. Children with poor functional outcomes at age 5 had worse educational outcomes age 7. Improvements in functional outcomes could enhance educational outcomes.

Background Children with persistent speech disorder (PSD) are at higher risk of difficulties with literacy, with some evidence suggesting an association with poorer educational attainment. However, studies to date have either used small clinical samples, which exclude children who have not been referred to clinical services, or relied on parent–teacher report of children's speech development. There is a need for an inclusive study to investigate the impact of PSD on educational outcomes using a population‐based sample and robust measures of speech development. Aim Using a large prospective UK population‐based study—the Avon Longitudinal Study of Parents and Children (ALSPAC)—this study investigated: (1) how children identified with PSD at age 8 years perform on educational attainment tests at ages 10–11 and 13–14 years in comparison with children without PSD; and (2) whether children identified with PSD at age 8 years are more likely to receive a label of special educational needs (SEN) in secondary school. Methods & Procedures We examined the data for 263 children with PSD and 6399 controls who had speech assessed at age 8 years in a research clinic. Educational attainment was measured using data from English school standard attainment tests. Data on SEN categorization were obtained between 11 and 13 years of age. Children with PSD and controls were compared using regression analyses adjusted for biological sex, maternal age, verbal, performance and full‐scale IQ. Outcomes & Results Children with PSD at age 8 years were more likely to achieve lower attainment scores at ages 10–11 years in English and mathematics and across all three subjects of English, mathematics and science at ages 13–14 years after controlling for biological sex and maternal education; score below target levels for English at both time points after controlling for verbal IQ, and at ages 13–14 years after controlling for performance IQ; and receive a label of SEN (typically for the category of cognition and learning needs or communication and interaction needs) in secondary school. Conclusions & Implications PSD identified at age 8 years is associated with poor educational attainment at ages 10–11 and 13–14 years in the core subjects of English, mathematics and science. Children with PSD at age 8 years are more likely to be identified with SEN at ages 11–13 years, particularly cognition and learning needs, and communication and interaction needs. We need to be aware of the potential for the long‐term impact of PSD on educational attainment in providing appropriate and effective support throughout school. What this paper addsWhat is already known on the subject Speech‐sound disorder is associated with reading and spelling difficulties, with some evidence to suggest that PSD is associated with a higher risk of literacy difficulties. Limited evidence also suggests that speech‐sound disorder may be associated with poorer educational attainment. However, studies to date have used small clinical samples or parent–teacher report of speech development and there is a need to determine whether the association is observed in larger and more inclusive population‐based samples. What this paper adds to existing knowledge This prospective, longitudinal study of a large community‐based sample of English children has shown that PSD is associated with poorer educational attainment at the end of primary school and at ages 13–14 years. Children with PSD are also more likely to be identified as having SEN in secondary school, especially communication and interaction needs but also including cognition and learning needs. What are the potential or actual clinical implications of this work? Understanding the long‐term implications of PSD on educational attainment highlights the importance of ongoing monitoring and support to enable children to reach their potential throughout primary and secondary school. The identification of children with a history of PSD during transition to secondary school will enable effective support to be put in place. The intervention for children with PSD should involve close collaboration between speech and language therapists and education professionals.

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 −8) that we pursued bioinformatically. We prioritized 5 novel gene regions with likely functional repercussions on neural pathways, including those which colocalized with differentially methylated regions in our sample. Polygenic risk scores for receptive language, expressive vocabulary, phonological awareness, phonological memory, spelling, and reading decoding associated with increasing clinical severity. In summary, neural-genetic influence on SSD is primarily multigenic and acts on genomic regulatory elements, similar to other neurodevelopmental disorders.

Objective: To provide comparison data on the Intelligibility in Context Scale (ICS) for a sample of 3-year-old English-speaking children born with any cleft type. Design: Questionnaire data from the Cleft Collective Cohort Study were used. Descriptive and inferential statistics were carried out to determine difference according to children’s cleft type and syndromic status. Participants: A total of 412 children born with cleft lip and/or palate whose mothers had completed the ICS when their child was 3 years old. Main Outcome Measure(s): Mothers’ rating of their children’s intelligibility using the ICS. Results: The average ICS score for the total sample was 3.75 ( sometimes-usually intelligible; standard deviation [SD] = 0.76, 95% CIs = 3.68-3.83) of a possible score of 5 ( always intelligible). Children’s speech was reported to be most intelligible to their mothers (mean = 4.33, SD = 0.61, 95% CIs = 4.27-4.39) and least intelligible to strangers (mean = 3.36, SD = 1.00, 95% CIs = 3.26-3.45). There was strong evidence ( P < .001) for a difference in intelligibility between children with cleft lip only (n = 104, mean = 4.13, SD = 0.62, 95% CIs = 4.01-4.25) and children with any form of cleft palate (n = 308, mean = 3.63, SD = 0.76, 95% CIs = 3.52-3.71). Children born with cleft palate with or without cleft lip and an identified syndrome were rated as less intelligible (n = 63, mean = 3.28, SD = 0.85, 95% CIs = 3.06-3.49) compared to children who did not have a syndrome (n = 245, mean = 3.72, SD = 0.71, 95% CIs = 3.63-3.81). Conclusions: These results provide preliminary comparative data for clinical services using the outcome measures recommended by the International Consortium for Health Outcomes Measurement.

Connected speech (CS) is an important component of child speech assessment in both clinical practice and research. There is debate in the literature regarding what size sample of CS is required to facilitate reliable measures of speech output. The aim of this study was to identify the minimum number of word tokens required to obtain a reliable measure of CS across a range of measures. Participants were 776 5-year-olds from a longitudinal community population cohort study (Avon Longitudinal Study of Parents and Children, ALSPAC). Children’s narratives from a story retell task were audio-recorded and phonetically transcribed. Automatic analysis of the transcribed speech samples was completed using an automated transcription and analysis system. Measures of speech performance extracted included: a range of profiles of percentage consonant correct; frequency of substitutions, omissions, distortions and additions (SODA); percentage of syllable and stress pattern matches; and a measure of whole word complexity (Phonological Mean Length of Utterance, pMLU). Statistical analyses compared these measures at different CS sample sizes in increments using averages and weighted moving averages, and investigated how measures performed between CS samples grouped into word tokens of at least 50, 75 and 100, and restricted to samples of 50–74, 75–99 and 100–125. Key findings showed that sample sizes of 75 word tokens and above showed minimal differences in most measures of speech output, suggesting that the minimum requirement for samples of CS is a word count of 75. The exception to this is in the case of pMLU and measures of substitutions and distortions when a word count of 100 is recommended.

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.

A child born with a cleft lip and palate will face 20 years or more of hospital care and surgery. This is a global problem with approximately 10 million people affected worldwide. Various models of care exist around the condition, and the best configurations of services within an economy need to be optimized. We provide examples of how centralized care can improve outcomes and provide an opportunity to establish national registries, and then emphasize the opportunities for building research platforms of relevance. The default of any cleft service should be to centralize care and enable cleft teams with a sufficient volume of patients to develop proficiency and measure the quality of outcomes. The latter needs to be benchmarked against the better centers in Europe. Two areas of concern for those with cleft are morbidity/mortality and educational attainment. These two issues are placed in context within the literature and wider approaches using population genetics. Orthodontists have always played a key role in developing these initiatives and are core members of cleft teams with major responsibilities for these children and their families., Highlights • Cleft lip and palate affects about 10 million people worldwide. • Centralised care can improve outcome and establish national registries. • Orthodontists have played a key role in developing these initiatives. • No strong evidence of an association between clefting and cancer. • Proper care can positively influence lives of children born with a cleft.

Introduction: Children born with a cleft palate ± lip are at risk of developing speech and language difficulties, which may require intervention from a speech and language therapist (SLT). To date, there is no strong evidence to support one approach to intervention over another, neither is it clear which approaches or methods of provision are commonly used. Objectives: To describe the range of speech and language therapy interventions being used with children born with cleft palate in the United Kingdom up to 5 years of age. To explore the different ways, interventions are being delivered. Design: A prospective study to conduct 9 semi-structured focus groups. Iterative content analysis was completed. Setting: Regional Cleft Lip and Palate Centers in the United Kingdom. Participants: Sixty-two speech and language therapy professionals from specialist cleft teams and community services. Results: Four main codes were identified: “intervention approaches,” “service delivery models,” “decision-making and rationale,” and “patient-centered care.” Participants frequently discussed how they adopt an eclectic style when delivering intervention, the importance of an individualized approach for each child and service delivery constraints, such as a lack of resources. Conclusion: Insight into the multitude of intervention approaches used by SLTs, aspects which influence their decision-making and the variability of service delivery models were gained. Uncertainty regarding which intervention approaches and methods for delivery are most effective provides rationale for future research, to improve the effectiveness of speech and language intervention for children with cleft palate ± lip.

BACKGROUND: As a growing paradigm of health research, trainee collaboratives can influence clinical practice through the generation of cost-effective multicenter audit and research projects. The aims of the present article are to outline and discuss the establishment of a multidisciplinary collaborative in the context of cleft lip and/or palate (CL/P).METHODS: The Cleft Multidisciplinary Collaborative (CMC) was formed in April 2016 under the overarching supervision of the National Institute for Health Research. Membership of the CMC is open to all members of the CL/P multidisciplinary team, who are encouraged to submit ideas for new research projects that will benefit clinical practice.RESULTS: To date, 48 clinical participants are involved in the CMC. These participants represent all 17 cleft teams from the United Kingdom and encompass a wide range of disciplines. The CMC has undertaken 2 major projects so far. The first involved collection of phenotype data to support a national cohort study. The second, still in progress, is a systematic review investigating factors associated with outcomes for velopharyngeal competence following cleft palate repair.CONCLUSIONS: The concept of a multidisciplinary collaborative in CL/P has been demonstrated through the generation of a United Kingdom-wide network of committed clinicians and researchers and the effective undertaking of 2 large research projects. As the CMC gathers momentum, it hopes to attract funding to support its activities, to promote more involvement from the allied health and nursing professions, to encourage a more ingrained research culture within the CL/P community, and to promote the wider ambition of a global collaborative.

Children’s speech development begins in infancy. The pattern of this development has been explored in studies over a number of years using a range of research methodology and approaches to investigation. A systematic review of the existing literature was carried out to determine the collective contribution of this literature to our understanding of early vocalizations and babbling through the period 9 to 18 months. Eight bibliographic databases were searched as well as the Cochrane library. Thirteen studies were identified for inclusion, which were mostly longitudinal observational case series. The review identified progressive increases in the complexity and volume of infants’ early vocalizations through the period. It also found a broad order of phonological acquisition. Although the studies in this review demonstrated marked individual variation, the review provides indicative patterns of development which can be used as a basis to explore relationships with later speech development in future studies.