Should left- and right-sided unilateral cleft lip and palate patients be grouped together when reporting the outcomes?

OBJECTIVES: Investigate associations between cleft laterality in patients with non-syndromic unilateral cleft lip and palate (UCLP) and oral-health, dental-arch, speech, audiological, psychological and nasolabial-aesthetic outcomes. METHODS: Secondary data analysis of the outcomes of 5-year-old children with non-syndromic complete UCLP identified from three studies: Cleft Collective (n = 155), Cleft Care UK (CCUK) (n = 266) and Clinical Standards Advisory Group (CSAG) study (n = 238). Outcome measures included occlusal assessment using the 5-year-old's index score, speech intelligibility rating using the CAPS-A Audit tool, audiological assessment using pure tone audiometry, nasolabial aesthetic assessment using the Asher-McDade tool, oral-health assessment using decayed, missing, filled teeth scores and parent-reported outcomes. Logistic regression with adjustment for age, sex and index-of-multiple-deprivation scores were performed. RESULTS: No differences were found in patient-reported outcomes between the left and right clefts in the Cleft Collective study. From the CCUK study, right clefts had poorer speech (n = 236; 95% CI 1.09, 3.42; and P = .03) and hearing outcomes (n = 211; 95% CI 1.03, 3.43; P = .04). In the CSAG study, patients with left clefts were more likely to be teased (n = 213; 95% CI 0.26, 0.85; and P = .01). CONCLUSION: Weak associations between cleft laterality, speech, hearing and psychological outcomes were found, however the findings were inconsistent across the studies. This study contributes to evidence of associations between laterality and outcomes in children born with UCLP.

Prevalence and Factors Associated with Behavioral Problems in 5-Year-Old Children Born with Cleft Lip and/or Palate from the Cleft Collective.

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

Speech sound disorder or DLD (phonology)? Towards a consensus agreement on terminology.

BACKGROUND: The 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. AIMS: The 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. METHODS: Following 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. CONCLUSIONS: Existing 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 would obviate the need for a Delphi process. WHAT THIS PAPER ADDS: What is already known on this subject Over nearly a hundred years, as our knowledge and understanding of speech sound disorder (SSD) has increased, so has the terminology that is used to describe those disorders. Current terminology not only describes subtypes of SSD but can also signpost us to effective interventions. With the publication, in 2017, of phase 2 of CATALISE a new term of 'developmental language disorder (DLD) (phonology)' was introduced with the unintentional consequence of challenging more specific descriptive terms for SSD. What this paper adds In the context of CATALISE and DLD (phonology), the history and nature of SSD terminology are reappraised. Building on the model of DLD developed in CATALISE, a tiered model that supports descriptive diagnosis and signposts intervention is proposed for discussion. Clinical implications of this study The proposed model of terminology for SSD provides descriptive and detailed labels that will support accuracy in differential diagnosis of developmental SSD by speech and language therapists. Furthermore, a decision-making tree for SSD demonstrates the pathway from diagnostic use of the terminology to the selection of evidence-based, effective interventions.

Current Surgical Practice for Children Born with a Cleft lip and/or Palate in the United Kingdom.

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 < .001) and a 5 to 7-day postoperative regime used more commonly for soft palatoplasties (P < .001). Perioperative steroids were used more commonly in palatoplasties than cheiloplasties (P < .001) but tranexamic acid use was equivalent (P = .73). 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.

Sidedness in Unilateral Orofacial Clefts: A Systematic Scoping Review.

OBJECTIVE: An overview of the literature relating to the sidedness of unilateral cleft lip with or without cleft palate to map current knowledge on the cause and impact of directional asymmetry. DESIGN: Scoping review with a systematic search of Medline and Embase from inception to May 2023. PATIENTS, PARTICIPANTS: Humans born with a left or right unilateral cleft lip with or without a cleft palate. MAIN OUTCOME MEASURES: Cleft sidedness as a co-occurrence, an outcome or an exposure. RESULTS: Forty studies were eligible for inclusion and confirmed the predilection for the occurrence of left sided cleft lips; 12 studies reported cleft sidedness co-occurring with another phenotype, 11 studies report sidedness as an outcome and 17 studies as an exposure. Phenotypes which were reported to co-occur with either left or right sided clefts included congenital dental anomalies, handedness and additional congenital anomalies. Variables investigated as a potential cause of left or right sided clefts as an outcome included chromosomal anomalies, genetic variants and environmental factors. Outcomes investigated in relation to cleft sidedness as an exposure included facial anatomical features, facial growth, educational attainment, functional and psychological characteristics. More studies showed worse outcomes in right sided clefts versus left sided clefts than vice versa, although studies were inconsistent, and a quality assessment was not performed. CONCLUSIONS: The field of cleft sidedness research is expanding and there are promising early findings to differentiate cause and outcome by sidedness of the cleft.

Patient Factors Influencing Speech Outcomes in Velopharyngeal Function Following Initial Cleft Palate Repair: A Systematic Review and Meta-Analysis.

OBJECTIVE: Identification of patient factors influencing velopharyngeal function for speech following initial cleft palate repair. DESIGN: A literature search of relevant databases from inception until 2018 was performed using medical subject headings and keywords related to cleft palate, palatoplasty and speech assessment. Following three stage screening data extraction was performed. SETTING: Systematic review and meta-analysis of relevant literature. PATIENTS/PARTICIPANTS: Three hundred and eighty-three studies met the inclusion criteria, comprising data on 47 658 participants. INTERVENTIONS: Individuals undergoing initial palatoplasty. MAIN OUTCOME MEASURES: Studies including participants undergoing initial cleft palate repair where the frequency of secondary speech surgery and/or velopharyngeal function for speech was recorded. RESULTS: Patient factors reported included cleft phenotype (95% studies), biological sex (64%), syndrome diagnosis (44%), hearing loss (28%), developmental delay (16%), Robin Sequence (16%) and 22q11.2 microdeletion syndrome (11%). Meta-analysis provided strong evidence that rates of secondary surgery and velopharyngeal dysfunction varied according to cleft phenotype (Veau I best outcomes, Veau IV worst outcomes), Robin Sequence and syndrome diagnosis. There was no evidence that biological sex was associated with worse outcomes. Many studies were poor quality with minimal follow-up. CONCLUSIONS: Meta-analysis demonstrated the association of certain patient factors with speech outcome, however the quality of the evidence was low. Uniform, prospective, multi-centre documentation of preoperative characteristics and speech outcomes is required to characterise risk factors for post-palatoplasty velopharyngeal insufficiency for speech. SYSTEMATIC REVIEW REGISTRATION: Registered with PROSPERO CRD42017051624.

A systematic review of early speech interventions for children with cleft palate.

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.

Maternal Cigarette Smoking and Cleft Lip and Palate: A Systematic Review and Meta-Analysis.

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

Changes in the Transverse Dimension of the Maxillary Arch of 5-Year-Olds Born With UCLP Since the Introduction of Nationwide Guidance.

OBJECTIVE: To determine whether the transverse dimensions of the maxillary arch of 5-year-old children with unilateral cleft lip and palate (UCLP) have changed following centralization of cleft services in the United Kingdom. DESIGN: Retrospective cross-sectional study. SETTING: Digital analysis of UCLP maxillary dental casts. PARTICIPANTS: All available maxillary dental casts from 5-year-old participants of the Clinical Standards Advisory Group (CSAG, N = 114) and Cleft Care UK (CCUK, N = 175) studies. INTERVENTIONS: Quantitative measurements of the intercanine width (ICW), intermolar width (IMW), and the distance from the midline to the greater and lesser side canine (GC/LC) and greater side and lesser side second primary molar (GE/LE). Degree measurements of the greater and lesser arch form angles, arch length, anterior palatal depth (APD), and posterior palatal depth were also measured. MAIN OUTCOME: Differences between the transverse dimensions of the maxillary arch for the CSAG and CCUK cohorts. RESULTS: In 5 (ICW, IMW, LC, LE, and APD) of the 11 measurements, there was a statistically significant difference between the CSAG and CCUK cohorts. In all of these, the CCUK values were greater than CSAG. CONCLUSIONS: There have been small but positive improvements for the transverse maxillary dimensions since centralization of the UK cleft service.

Early Communication Behaviors in Infants With Cleft Palate With and Without Robin Sequence: A Preliminary Study.

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.

Exploring the Relationship Between Palatal Cleft Type and Width With the Use of Relieving Incisions in Primary Repair.

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.

Maternal Stressful Life Events During the Periconceptional Period and Orofacial Clefts: A Systematic Review and Meta-Analysis.

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.

Making the case for the collection of a minimal dataset for children with speech sound disorder.

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.

Speech input processing in children born with cleft palate: A systematic literature review with narrative synthesis.

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.

Educational outcomes associated with persistent speech disorder.

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 adds What 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.

An Exploratory Study of Speech and Language Therapy Intervention for Children Born With Cleft Palate ± Lip.

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.

Intelligibility in 3-Year-Olds With Cleft Lip and/or Palate Using the Intelligibility in Context Scale: Findings from the Cleft Collective Cohort Study.

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.

Educational Attainment of Children Born with Unilateral Cleft Lip and Palate in the United Kingdom.

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.

The Relationship between Feeding and Non-Nutritive Sucking Behaviours and Speech Sound Development: A Systematic Review.

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.

How many words make a sample? Determining the minimum number of word tokens needed in connected speech samples for child speech assessment.

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.