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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.

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.

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.

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.

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.

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.

A feasibility study of The Fluency Trust Residential Course (FTRC) for adolescents who stutter was conducted. The study aimed to measure key areas of a feasibility trial, for example, recruitment and retention, outcome measure completion, outcome measure reliability, and acceptability of the intervention to inform future research into the FTRC.Quantitative and qualitative methods were used. Participants were 23 adolescents (12-17 years), 23 parents and 2 Speech-Language Pathologists (SLPs) from the FTRC. Data collection included: outcome measure collection via a pre-test post-test quasi-experimental design (including two baseline measures), intervention fidelity checklists, semi-structured interviews with adolescents to explore acceptability of the intervention and semi-structured interviews with SLPs to explore their experiences of research participation and views on a future trial.Recruitment, retention and outcome measure completion levels were all 100%. Intervention fidelity was 95% and there were no adverse events. Outcome measures showed good test- re-test reliability: Progress Questionnaire Child Intraclass Correlation Coefficient (ICC) = 0.87 (95% CI = 0.69-0.94 sig 0.001) and Progress Questionnaire Parent ICC = 0.88 (95% CI = 0.70-0.95 sig 0.001). Descriptive statistics showed that group medians and means of all outcome measures shifted in a positive direction between pre and post-tests (9 weeks follow-up). Twenty-five percent of young people showed changes on the Progress Questionnaire Child that were above the minimal important difference. Seventy-five percent of parents showed changes on the Progress Questionnaire Parent that were above the minimal important difference. Acceptability of the intervention by adolescents was high. SLPs reported participation was manageable and they were pleased to be part of the research.Quantitative and qualitative data suggest that a future definitive trial of the FTRC is indicated after additional development work and feasibility testing. Recommendations for further research are included.

Children with cleft palate are at high risk of both conductive hearing loss and cleft speech characteristics (CSCs) yet there is limited research to understand whether hearing loss impacts speech development in this population by contributing to the development of CSCs.This study used data from a large national cohort study in the UK (The Cleft Collective) to explore the relationship between those with a history of diagnosed hearing loss and presence of CSCs in children with cleft palate (+/- cleft lip) aged between 18 and 24 months.Speech and Language Therapists (SLTs) provided uniformed information from assessment for 123 participants who had been recruited to the Cleft Collective study. History of diagnosed hearing loss, intervention for hearing loss, and presence of CSCs were reported. A consonant inventory for each participant was completed. Statistical analysis of relationships between hearing loss and CSCs and analysis of consonant inventories was undertaken to provide information related to speech acquisition and its relationship with hearing.There was a statistically significant relationship between history of diagnosed hearing loss and presence of CSCs (p 0.05). Analysis of the consonant inventories highlighted that children with diagnosed hearing loss used fewer oral consonants compared to those with normal hearing (p 0.05) CONCLUSION: This study provides some evidence that a conductive hearing loss can affect speech in children with cleft palate resulting in CSCs developing. Therefore, children with cleft palate should be closely monitored by audiology from birth to ensure that hearing is optimised for speech and language development. SLT services should also closely monitor the speech development of those children where a hearing loss has been identified, so that early intervention can be provided if appropriate. Continued data collection, with a large sample of children, will provide additional evidence regarding how this hearing loss is best managed. It will also allow increased knowledge of the long term impact of conductive hearing loss on speech development in children with cleft palate.

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

BACKGROUND: Linguistic comprehension and narrative skills encapsulate a complex array of grammatical and semantic skills that underpin complex reading comprehension processes. However, most research in this area has focused on children with reading difficulties and not on typically developing children. Also the research has mostly focused on short-term effects of these skills on reading during the primary school years. Therefore, it remains unclear what specific role linguistic comprehension and narrative skills play in typically developing children's reading beyond the primary school years.AIMS: With this 9-year prospective longitudinal study, we sought to clarify the independent effects of linguistic comprehension and narrative skill (at 5 years of age) on children's reading ability at 10 and 14 years of age.SAMPLE: We examined the data from 716 children (MAge = 67 months, SD = 2.13 months), which were drawn from a major population cohort study, the Avon Longitudinal Study of Parents and Children.METHODS: Children's language skills were assessed at 5 and word reading and reading comprehension skills at 10 years of age. The reading achievement scores at 14 years of age were based on national curriculum test results.RESULTS: Linguistic comprehension and narrative skills at 5 years of age made unique and direct contributions to reading comprehension skills and reading achievement after accounting for general cognitive ability, memory, phonological skills, and mother's education. Moreover, listening comprehension predicted reading achievement even when prior reading skills were taken into account.CONCLUSIONS: Linguistic comprehension and narrative skills are related but distinct oral language skills that continue to influence children's reading development beyond the primary school years.

Recent studies report prevalence, phenotype, and persistence findings for a paediatric motor speech disorder in addition to childhood dysarthria and childhood apraxia of speech termed Speech Motor Delay (SMD). The aim of the present study was to determine if there is a frequent acoustic sign of SMD, with implications for theory, assessment, and treatment. We examined the frequency of 19 acoustic signs of SMD in audio recordings of continuous speech and word-imitation tasks in three groups of speakers with SMD: 50 children (mean age 5.1 years) with idiopathic Speech Delay (SD) from 6 USA cities; 87 children, adolescents, and adults with eight types of complex neurodevelopmental disorders; and 9 children (mean age 8.8 years) with persistent idiopathic SD from a population-based study of children in the South West of England. The 19 acoustic signs of imprecise or unstable speech, prosody, and voice were standardized on typical speakers of the appropriate dialect. The criterion for a frequent acoustic sign was that it occurred in at least 50% of participants with SMD in each of the three groups. Findings indicated that lengthened mid-vowels and diphthongs was the one sign that met criteria, occurring in 64.4% of the 146 participants with SMD, including 71% of the 87 participants with complex neurodevelopmental disorders. Findings are interpreted to support the potential of this acoustic sign, and possibly several others associated with temporal dimensions of speech sound development, to inform explication of the neuromotor substrates of SMD.

The work reported in this special issue of 'Folia Phoniatrica et Logopaedica', supported by the Child Speech Committee of the International Association of Logopedics and Phoniatrics (IALP), shows how transcription has changed over the last few decades and brings readers up to date with the state of the art. Transcription is a tool which is integral to the practice of speech-language pathologists (SLPs). Being able to hear what individuals say is useful, but only by documenting this, can it be determined whether change has occurred over time or in response to intervention. It is through close observation of the transcription that you can identify patterns of ability and error and use this information to plan clinical management. After a thorough review on the history of the transcription of children's speech it has been found that the emphasis within this special topic issue lies upon cross-linguistic transcription.

Purpose The aim of this tutorial is to support speech-language pathologists (SLPs) undertaking assessments of multilingual children with suspected speech sound disorders, particularly children who speak languages that are not shared with their SLP. Method The tutorial was written by the International Expert Panel on Multilingual Children's Speech, which comprises 46 researchers (SLPs, linguists, phoneticians, and speech scientists) who have worked in 43 countries and used 27 languages in professional practice. Seventeen panel members met for a 1-day workshop to identify key points for inclusion in the tutorial, 26 panel members contributed to writing this tutorial, and 34 members contributed to revising this tutorial online (some members contributed to more than 1 task). Results This tutorial draws on international research evidence and professional expertise to provide a comprehensive overview of working with multilingual children with suspected speech sound disorders. This overview addresses referral, case history, assessment, analysis, diagnosis, and goal setting and the SLP's cultural competence and preparation for working with interpreters and multicultural support workers and dealing with organizational and government barriers to and facilitators of culturally competent practice. Conclusion The issues raised in this tutorial are applied in a hypothetical case study of an English-speaking SLP's assessment of a multilingual Cantonese- and English-speaking 4-year-old boy. Resources are listed throughout the tutorial.

ObjectivesTo investigate centre-level variation in speech intervention and outcome and factors associated with a speech disorder in children in Cleft Care UK (CCUK).Setting and Sample PopulationTwo hundred and sixty-eight 5-year-old British children with non-syndromic unilateral cleft lip and palate recruited to CCUK.Materials and MethodsCentre-based therapists undertook audio-video recordings. Perceptual analysis was undertaken using the CAPS-A tool. Speech outcomes were based on structural and articulation scores, and intelligibility/distinctiveness. Between-centre variation in treatment and outcomes were examined using multilevel models. These models were extended to estimate the association between a range of factors (hearing loss, speech intervention, fistula, secondary speech surgery for velopharyngeal insufficiency, socio-economic status, gender, and parental happiness with speech) and speech outcomes.ResultsThere was centre-level variation in secondary speech surgery, speech intervention, structure and intelligibility outcomes. Children with a history of speech intervention had a lower odds of poor intelligibility/distinctiveness, 0.1 (95% CI: 0.0-0.4). Parental concern was associated with a higher odds of poor intelligibility/distinctiveness, 13.2 (95% CI: 4.9-35.1). Poor speech outcomes were associated with a fistula, secondary speech surgery and history of hearing loss.ConclusionsWithin the centralized service there is centre-level variation in secondary speech surgery, intervention and speech outcomes. These findings support the importance of early management of fistulae, effective management of velopharyngeal insufficiency and hearing impairment, and most importantly speech intervention in the preschool years. Parental concern about speech is a good indicator of speech status.

ObjectivesTo summarize and discuss centre-level variation across a range of treatment and outcome measures and examine individual and ecological determinants of outcome in children in Cleft Care UK (CCUK).Setting and sample populationTwo hundred and sixty-eight 5-year-old British children with non-syndromic unilateral cleft lip and palate (UCLP) recruited to CCUK and treated within a centralized service.Materials and MethodsChildren had a range of treatment and outcome measures collected at a 5-year audit clinic. These outcomes included dento-alveolar arch relationships from study models, measures of facial appearance from cropped photographs, hearing loss from audiological assessment, speech from speech recordings, self-confidence and strengths and difficulties from parental self-report. Data were collected on educational attainment at age 7 using record linkage. Centre variation was examined using hierarchical regression and associations between variables were examined using logistic or poisson regression.ResultsThere was centre-level variation for some treatments (early grommet placement, fitting of hearing aids, fluoride treatment, secondary speech surgery and treatment for cleft speech characteristics) and for some outcomes (intelligibility of speech). Hearing loss was associated with a higher risk of poor speech while speech therapy was associated with a lower risk of poor speech. Children had high levels of caries but levels of preventative treatment (fluoride varnish and tablets) were low.ConclusionsFurther improvements to and monitoring of the current centralized model of care are required to ensure the best outcomes for all children with cleft lip and palate.

As part of a large-scale study of children's development, 7390 children were assessed on a range of speech tasks. These were used to identify three groups of children with speech errors within the sample: persistent speech disorder (PSD); speech errors but below the threshold for classification as persistent speech disorder (non-PSD); and common clinical distortions only (CCD). These three groups were compared on demographic factors, performance on IQ, non-word repetition, and diadochokinetic tests. Findings showed that the PSD group and the non-PSD group were most similar for gender, socio-economic status, IQ, and non-word repetition. In the diadochokinetic tasks, the PSD group and the CCD groups were more similar. Implications for these findings in terms of clinical practice are discussed.

Background Some childhood speech and language impairments precede psychosis but it is not clear whether they also precede adolescent psychotic experiences and whether this association is specific to psychotic experiences. Methods Pragmatic language and expressive speech and language (parent-assessed using the Children's Communication Checklist) at age 9 and psychotic experiences and depression at ages 12 and 18 were investigated in 7659 participants from the Avon Longitudinal Study of Parents and Children. Associations were investigated using multivariate modelling. Results Poorer pragmatic language at 9 years was associated with psychotic experiences at both ages (12 years OR 1.22, 95% CI 1.11, 1.34; 18 years OR 1.25, 95% CI 1.10, 1.41) but only with depression at 18 years (OR 1.10, 95% CI 1.00, 1.22). Poorer expressive speech and language ability was not associated with psychotic experiences or depression at either age. There was evidence that pragmatic language was specifically associated with psychotic experiences at age 12 but no evidence that the strength of any of the associations changed over time. Conclusions Deficits in pragmatic language precede early and late adolescent psychotic experiences and early adolescent depression. Interventions aimed at helping children improve pragmatic language skills may reduce the incidence of adolescent psychopathology and associated psychological disorder and dysfunction later in life.

Purpose The purpose of this study was to determine prevalence and predictors of persistent speech sound disorder (SSD) in children aged 8 years after disregarding children presenting solely with common clinical distortions (i.e., residual errors). Method Data from the Avon Longitudinal Study of Parents and Children (Boyd et al., 2012) were used. Children were classified as having persistent SSD on the basis of percentage of consonants correct measures from connected speech samples. Multivariable logistic regression analyses were performed to identify predictors. Results The estimated prevalence of persistent SSD was 3.6%. Children with persistent SSD were more likely to be boys and from families who were not homeowners. Early childhood predictors identified as important were weak sucking at 4 weeks, not often combining words at 24 months, limited use of word morphology at 38 months, and being unintelligible to strangers at age 38 months. School-age predictors identified as important were maternal report of difficulty pronouncing certain sounds and hearing impairment at age 7 years, tympanostomy tube insertion at any age up to 8 years, and a history of suspected coordination problems. The contribution of these findings to our understanding of risk factors for persistent SSD and the nature of the condition is considered. Conclusion Variables identified as predictive of persistent SSD suggest that factors across motor, cognitive, and linguistic processes may place a child at risk.

A focus group of seven speech and language therapists (SLTs) who work with children with phonological difficulties was invited to share their views on their current ways of working. Qualitative data was generated through discussion of two video-taped recordings of children with phonological difficulties and a series of brainstorming tasks. The results indicate the use of an electric approach, a mixture of theories rather than a single one. Similarities and differences between SLTs are identified. Implications for the development of clinical guidelines and the investigation of effectiveness are discussed.

Children who are bilingual and have speech sound disorder are likely to be under-referred, possibly due to confusion about typical speech acquisition in bilingual children.To investigate what is known about the impact of bilingualism on children's acquisition of speech in English to facilitate the identification and treatment of bilingual children with speech sound disorder.A systematic review of studies from the last 50 years was conducted. Studies investigating speech acquisition in bilingual infants and children (where one language was English) were identified through searching seven electronic databases, bibliographies of relevant articles and e-mailing authors. Sixty-six studies investigating bilingual speech production met inclusion criteria, with 53 describing typically developing children and 13 describing children with speech sound disorder. The 66 studies were analysed thematically and summarized in terms of methods, key findings and underlying theories.There was limited evidence to suggest that bilingual children develop speech at a slower rate than their monolingual peers; however, there was evidence for qualitative differences and increased variation in speech production. Nearly all studies provide evidence for transfer between the two phonological and language structures, although the amount of transfer varied between studies. There was evidence of positive and negative transfer of features from the dominant language (L1) to the second language (L2) as well as from L2 to L1. Positive transfer became more evident with increased age and length of exposure to a second language. More recently researchers have moved away from investigating whether there are one or two phonological systems and accept that there are two systems that interact. Interest has shifted to examining how phonological systems interact and to identifying factors that influence interactions. The review revealed a number of inconsistencies in the findings of studies due to differences in methodology, languages investigated and degree of language exposure. Overall, measurement issues were addressed well but most studies provided limited sample information about language experience, schooling and socio-economic status.There are differences in speech sound acquisition between monolingual and bilingual children in terms of rate and patterns of error, with both positive and negative transfer occurring in bilingual children.

A major challenge for the speech-language pathology profession in many cultures is to address the mismatch between the “linguistic homogeneity of the speech-language pathology profession and the linguistic diversity of its clientele” ( Caesar & Kohler, 2007 , p. 198). This paper outlines the development of the Multilingual Children with Speech Sound Disorders: Position Paper created to guide speech-language pathologists’ (SLPs’) facilitation of multilingual children's speech. An international expert panel was assembled comprising 57 researchers (SLPs, linguists, phoneticians, and speech scientists) with knowledge about multilingual children's speech, or children with speech sound disorders. Combined, they had worked in 33 countries and used 26 languages in professional practice. Fourteen panel members met for a one-day workshop to identify key points for inclusion in the position paper. Subsequently, 42 additional panel members participated online to contribute to drafts of the position paper. A thematic analysis was undertaken of the major areas of discussion using two data sources: (a) face-to-face workshop transcript (133 pages) and (b) online discussion artifacts (104 pages). Finally, a moderator with international expertise in working with children with speech sound disorders facilitated the incorporation of the panel's recommendations. The following themes were identified: definitions, scope, framework, evidence, challenges, practices, and consideration of a multilingual audience. The resulting position paper contains guidelines for providing services to multilingual children with speech sound disorders ( http://www.csu.edu.au/research/multilingual-speech/position-paper ). The paper is structured using the International Classification of Functioning, Disability and Health: Children and Youth Version ( World Health Organization, 2007 ) and incorporates recommendations for (a) children and families, (b) SLPs’ assessment and intervention, (c) SLPs’ professional practice, and (d) SLPs’ collaboration with other professionals. Learning outcomes: Readers will 1. recognize that multilingual children with speech sound disorders have both similar and different needs to monolingual children when working with speech-language pathologists. 2. Describe the challenges for speech-language pathologists who work with multilingual children. 3. Recall the importance of cultural competence for speech-language pathologists. 4. Identify methods for international collaboration and consultation. 5. Recognize the importance of engaging with families and people within their local communities for supporting multilingual children in context.

Speech disorder that continues into middle childhood is rarely studied compared with speech disorder in the early years. Speech production in single words, connected speech and nonword repetition was assessed for 7390 eight-year-old children within the Avon Longitudinal Study of Parents and Children (ALSPAC). The majority ( n = 6399) had typical speech and 50 of these children served as controls. The remainder were categorised as using common clinical distortions only (CCD, n = 582) or speech difficulties (SDiff, n = 409). The samples from the CCD children were not analysed further. Speech samples from the SDiff and the control children were transcribed and analysed in terms of percentage consonants correct, error type and syllable structure. Findings were compared with those from children in the Shriberg et al. (1997) lifespan database ( n = 25). The 8-year-old children from ALSPAC in the SDiff and control groups achieved similar speech accuracy scores to the 8-year-old children in the lifespan database. The SDiff group had consistently lower scores than the ALSPAC control group, with the following measures most clearly differentiating the groups: single word task (percentage of substitutions and distortions), connected speech task (percentage of vowels correct (PVC), percentage of omission of singletons and entire clusters, and stress pattern matches), nonword repetition task (PVC, percentage of entire clusters omitted, percentage of distortions, and percentage of stress pattern matches). Connected speech and nonword samples provide useful supplementary data for identifying older children with atypical speech. Learning outcomes: The reader will recognize the methods used to identify speech characteristics in a large scale population study. They will describe how measures of speech accuracy in connected speech compare with the Shriberg et al. (1997) lifespan database. The reader will also recall information on how typically and atypically developing children differ on a range of measures across different types of speech sample.

The move towards an inclusive model of education presents teachers with the difficulty of differentiating the curriculum for children with speech, language and communication impairments. This paper focuses on the ‘WiSaLT Curriculum Appendix’ - a tool which can be used by teachers and speech and language therapists to help such children access the mainstream curriculum and to promote improvement in their language and communication skills. As well as highlighting potential areas of difficulty within each attainment target for key stage one, the appendix guides users to specific strategies and activities. Thus the speech and language therapist and teacher can identify which attainment targets might prove problematic for any one child and also have access to ideas which can help.