A meta-analysis of the relationship between speech and language development in children with nonsyndromic cleft palate with or without cleft lip.

OBJECTIVE: Children with nonsyndromic cleft palate with or without cleft lip are at risk of speech production and language delays. In typical development, a strong relationship exists between speech and expressive language development. However, the understanding of this relationship in children with nonsyndromic cleft palate with or without cleft lip is incomplete. The objective of this study was to determine the average correlation between consonant inventory and two expressive language skills in children with NSCP ± L. DESIGN: The study used a random effects meta-analyses design. Articles were included by searching PubMed, APA PsycINFO, and ERIC. MAIN OUTCOME MEASURES: Correlations between consonant inventory and (1) expressive vocabulary and (2) mean length of utterance. RESULT: Based on eight studies representing 187 children with nonsyndromic cleft palate with or without cleft lip, consonant inventory was positively correlated with expressive vocabulary size (k = 8, z = 0.74, SE = .16, p < .001, [0.424, 1.052]). Using data from four studies, representing 83 children with nonsyndromic cleft palate with or without cleft lip, consonant inventory had a positive nonsignificant correlation with mean length of utterance (k = 4, z = 0.513, SE = 0.293, p = .081, [-0.063, 1.088]). CONCLUSION: Results support a connection between consonant inventory and expressive vocabulary size in children with nonsyndromic cleft palate with or without cleft lip across four languages. Small consonant inventories are often associated with expressive language delays.

Socioeconomic Deprivation Detrimentally Influences Language Outcomes in Toddlers With Cleft Palate.

PURPOSE: The purpose of this study was to examine the influence of socioeconomic deprivation on language and developmental outcomes in toddlers with cleft palate with or without cleft lip (CP ± L). Other factors known to influence language outcomes were also considered, including home language history, history of hearing problems, syndromic diagnoses, and sex. METHOD: A multicenter, cross-sectional study design was conducted. Data for 566 16-month-old toddlers with CP ± L were collected from 17 outpatient cleft palate clinics located throughout the United States. Outcome measures included the MacArthur-Bates Communicative Development Inventories, LENA Developmental Snapshot, age at first word as reported by the caregiver, and the Ages and Stages Questionnaires-Third Edition (ASQ-3). Multivariable linear or logistic regression was used to determine the influence of socioeconomic deprivation, as measured by the Area Deprivation Index, on language and developmental outcomes. RESULTS: Greater socioeconomic deprivation significantly predicted poorer language outcomes in toddlers with CP ± L, including receptive vocabulary words (p = .02), expressive vocabulary words (p = .02), and late-developing gestures (p = .02). Additionally, toddlers from less affluent neighborhoods produced their first words significantly later than their counterparts living in more affluent areas (p < .01). Lower maternal education levels predicted significantly increased risk for problem solving delays (p < .01), and patients with subsidized insurance were at significantly increased risk for personal-social delays on the ASQ-3 (p < .01). CONCLUSIONS: Children with CP ± L are susceptible to developmental delays associated with socioeconomic deprivation. These findings have implications for identifying a child's individual risk factors for developmental language disorders when conducting speech-language assessments. Future study should examine how inequities in care can be mitigated and addressed.

Feeding Management and Palate Repair Timing in Infants with Cleft Palate with and without Pierre Robin Sequence: A Multisite Study.

OBJECTIVES: Compare the feeding management practices in infants with cleft palate with and without Pierre Robin sequence (PRS) and determine if specific feeding difficulties or interventions predict delayed palate repair. DESIGN: Retrospective cross-sectional study. SETTING: Seventeen cleft palate teams contributed data. PATIENTS: 414 infants were included in this study: 268 infants with cleft palate only and 146 infants with cleft palate and PRS. PROCEDURES: Data were collected via parent interview and electronic health records. MAIN OUTCOME MEASURES: Outcomes for the primary objective included categorical data for: history of poor growth, feeding therapy, milk fortification, use of enteral feeding, and feeding difficulties. The outcome for the secondary objective was age in months at primary palate repair. RESULTS: Infants with PRS had a significantly higher prevalence of feeding difficulties (81% versus 61%) and poor growth (29% versus 15%) compared to infants with cleft palate only. Infants with PRS received all feeding interventions-including feeding therapy, milk fortification, and enteral feeding-at a significantly higher frequency. Infants with PRS underwent primary palate repair at a mean age of 13.55 months (SD = 3.29) which was significantly (P < .00001) later than infants with cleft palate only who underwent palate repair at a mean age of 12.05 months (SD = 2.36). Predictors of delayed palate repair included diagnosis of PRS as well as Hispanic ethnicity and a history of poor growth. CONCLUSIONS: These findings can be used to establish clinical directives focused on providing early, multimodal feeding interventions to promote optimal growth and timely palate repair for infants with PRS.

Enhanced milieu teaching with phonological emphasis: A pilot, telepractice study for parent training in early intervention.

PURPOSE: This pilot study evaluated enhanced milieu teaching with phonological emphasis (EMT + PE) parent training intervention delivered through telepractice and its impact on parent implementation, child speech outcomes, and child language outcomes. METHOD: A multiple baseline design across behaviours was used to assess response to parent training and child outcomes. The intervention was delivered to four parents and their young children with repaired cleft palate. Speech production, receptive language, and expressive language were assessed prior to intervention. EMT + PE strategies including matched turns, modelling and expansions, and prompting/speech recasting were taught to parents using the teach-model-coach-review model. Social validity questionnaires were administered pre- and post-intervention to assess parents' rating of competence and confidence for managing their child's early development. Reliability and procedural fidelity were conducted. RESULT: All parents increased their use of modelling and expansions, and prompting and speech recasting in response to training, as evidenced by large effect sizes. Children also responded positively to these strategies by increasing percentage of consonants correct (PCC) and target vocabulary, although PCC gains were less than observed in a prior hybrid telepractice and face-to-face intervention. Social validity measures indicated parents increased their confidence to provide intervention for their children. CONCLUSION: This pilot study indicated that effective parent training in EMT + PE strategies can be delivered through telepractice.

Standardization of Videorecorded Speech Samples for Children with Cleft Palate: Methods to Facilitate High-Quality Speech Outcomes Data Collection.

BACKGROUND: Collection of high-quality videorecorded speech samples is essential for speech outcomes research. SOLUTION: Cleft palate team SLPs were trained to collect standard videorecorded speech samples in the clinic setting across 20 sites in North America. Standard training and equipment was provided. WHAT WE DID THAT IS NEW: Quality management procedures were developed and utilized to verify video quality and protocol adherence. Over 97% of speech samples collected by trained SLPs met defined quality standards.

Speech Production Errors in Children With Cleft Palate With or Without Cleft Lip.

PURPOSE: The purpose of this study was to describe the frequency of speech production errors in children with cleft palate with or without cleft lip (CP±L) and explore characteristics related to speech production errors. METHOD: Fifty-six children with nonsyndromic CP±L between the ages of 4;0 and 7;11 (years;months) were included in this study. The children's audio-recorded production of a sentence repetition task was transcribed using narrow transcription and coded for speech error type. RESULTS: Children used, on average, 18 speech errors during the sentence repetition task that sampled 59 phoneme targets. On average, phonological errors were used most frequently, with nine errors per sample, followed by anterior oral speech errors at four errors per sample, and non-oral compensatory errors at three errors per sample. Individual-level characteristics including age and cleft type were related to frequency of phonological errors and anterior oral speech errors, respectively. One treatment-level characteristic, hypernasality, was related to use of non-oral compensatory errors and passive speech errors. CONCLUSIONS: This study identified the most frequent speech production errors as phonological errors, followed by anterior oral speech errors and non-oral compensatory errors. Individual-level and treatment-level characteristics were related to speech production errors. Future research should explore additional characteristics that may influence use of speech production errors. Clinically, this study adds information regarding speech error types that should be monitored throughout cleft care, including phonological errors that were most prevalent among this sample. SUPPLEMENTAL MATERIAL: https://doi.org/10.23641/asha.22044095.

Reading Development in Children With Nonsyndromic Cleft Palate With or Without Cleft Lip: Meta-analysis and Systematic Review.

OBJECTIVE: We conducted a meta-analysis and systematic review of literature comparing pre-reading and general reading in school-age children with nonsyndromic cleft palate with or without cleft lip (NSCP/L) to their peers without NSCP/L. METHODS: Our literature search identified 1238 possible records. After screening we identified 11 samples for inclusion for systematic review and eight for meta-analysis. We compared 292 children with NSCP/L to 311 peers for 23 pre-reading effect sizes and 17 general reading effect sizes (EFg). We conducted a random-effects metaregression using robust variance estimation. RESULTS: On average school-age children with NSCP/L scored lower on pre-reading (EFg = -0.36) and general reading measures (EFg = -0.38) compared to their peers. We conducted post-hoc analyses on phonological awareness and word decoding effect sizes; children with NSCP/L performed lower on phonological awareness (EFg = -0.22) and word decoding (EFg = -0.39) compared to their peers. There was weak evidence that hearing status and/or speech-language functioning might moderate reading development. There was limited evidence that age or socioeconomic status moderated reading development. However, samples did not consistently report several characteristics that were coded for this project. CONCLUSIONS: Our findings suggest that school-age children with NSCP/L have persistent reading problems. Further research is needed to explore reading development in children with NSCP/L, as well as the relationships among hearing, speech, language, and reading development.

Enhanced Milieu Teaching with Phonological Emphasis: A Pilot Telepractice Parent Training Study for Toddlers with Clefts.

OBJECTIVE: the purpose of this study was to evaluate the effects of training caregivers to use intervention strategies from the Enhanced Milieu Teaching with Phonological Emphasis (EMT + PE) program, delivered via telepractice, and to examine the effects on child speech and language outcomes for children with repaired cleft lip +/- palate (CL/P). DESIGN: A multiple baseline within subject design across parent behaviors was replicated across three participating dyads. A pre-post intervention comparison was provided with a non-cleft twin. PARTICIPANTS: Three mother-child dyads participated in this study. Children ranged in age from 21 to 27 months at the beginning of the study and all had a diagnosis of CL/P. A noncleft twin without CL/P was assessed pre- and post-intervention to provide a normative comparison. RESULTS: Parents demonstrated a positive intervention effect by substantially increasing their use of EMT + PE intervention strategies during telepractice intervention sessions (Tau 0.675 to 1.1333). Following the conclusion of intervention, parents were able to maintain their use of strategies once direct coaching had been discontinued. Children demonstrated increased talking rate, improved speech production and expanded expressive vocabulary measures over the course of intervention. Speech and language development of a child without cleft palate was provided as a comparison. CONCLUSIONS: Parents were trained through telepractice to effectively deliver EMT + PE speech and language facilitation strategies that resulted in increased language and speech outcomes for their children with CL/P.

Effects of a naturalistic intervention on the speech outcomes of young children with cleft palate.

Purpose: The purpose of this study was to investigate the extent to which a naturalistic communication intervention, Enhanced Milieu Teaching with Phonological Emphasis (EMT + PE) improved the speech outcomes of toddlers with cleft palate with or without cleft lip (CP ± L)Method: This study was a stratified randomised controlled trial.Setting: Treatment was delivered in a university clinic by a trained speech-language pathologist.Thirty children aged between 15 and 36 months (M = 25) with nonsyndromic CP ± CL and typical cognitive development were randomly assigned to a treatment (EMT + PE) or business as usual comparison condition.Participants in the EMT + PE treatment group received 48, 30-min sessions, over a 6-month period. Fidelity of treatment was high across participants.The primary outcome measures were percent consonants correct (PCC), consonant inventory, compensatory articulation errors, and nasal emission.Result: Regression analyses controlling for pre-intervention child characteristics were conducted for PCC and consonant inventory. Intervention was not a significant predictor of post-intervention outcome. Words per minute differentiated the children who benefitted from the intervention from those who did not. Reduction in compensatory errors and nasal emission occurred in both groups but to a greater degree in the EMT + PE group.Conclusion: EMT + PE is a promising early speech intervention for young children with CP ± L, especially for children with higher rates of word use.

The Effectiveness of Palate Re-Repair for Treating Velopharyngeal Insufficiency: A Systematic Review and Meta-Analysis.

BACKGROUND: Palate re-repair has been proposed as an effective treatment for velopharyngeal insufficiency (VPI) with a low risk of obstructive sleep apnea (OSA). The authors conducted a systematic review and meta-analysis to determine the proportion of patients achieving normal speech resonance following palate re-repair for VPI, the proportion developing OSA, and the criteria for patient selection that are associated with increased effectiveness. METHODS: PubMed, Embase, and Scopus were searched from inception through April 2018 for English language articles evaluating palate re-repair for the treatment of VPI in patients with a repaired cleft palate. Inclusion criteria included reporting of hypernasality, nasal air emission, nasometry, additional VPI surgery, and/or OSA outcomes. Meta-analysis was conducted using random effects models. Risk of bias was assessed regarding criteria for patient selection, blinding of outcome assessors, and validity of speech assessment scale. RESULTS: Eighteen studies met inclusion criteria. The incidence of achieving no consistent hypernasality follow palate re-repair was 61% (95% confidence interval [CI]: 44%-75%). The incidence of additional surgery for persistent VPI symptoms was 21% (95% CI: 12%-33%). The incidence of OSA was 28% (95% CI: 13%-49%). Criteria for selecting patients to undergo re-repair varied, with anterior/sagittal position of palatal muscles (33%) and small velopharyngeal gap (22%) being the most common. No specific patient selection criteria led to superior speech outcomes (P = .6572). CONCLUSIONS: Palate re-repair achieves normal speech resonance in many but not all patients with VPI. Further research is needed to identify the specific examination and imaging findings that predict successful correction of VPI with re-repair.

Early Speech and Language Development in Children With Nonsyndromic Cleft Lip and/or Palate: A Meta-Analysis.

Objective The aim of the study was to conduct a meta-analysis of research examining the early speech and language functioning of young children, birth to age 8;11 (years;months), with nonsyndromic cleft lip and/or palate (NSCL/P) compared to their peers without NSCL/P. Method We conducted a random-effects metaregression using 241 effect sizes from 31 studies comparing 955 young children with NSCL/P to 938 typically developing peers on measures of speech and language functioning. Moderators were sample characteristics (i.e., age, cleft type, publication year, and study location) and measurement characteristics (i.e., speech sample material, language modality and domain, and assessment type). Results Young children with NSCL/P scored significantly lower on measures of speech and language compared to children without NSCL/P. Children with NSCL/P had smaller consonant inventories (standardized mean difference effect size [ESg] = -1.24), less accurate articulation (ESg = -1.13), and more speech errors (ESg = 0.93) than their peers. Additionally, children with NSCL/P had poorer expressive (ESg = -0.57) and receptive (ESg = -0.59) language skills than their peers. Age and assessment type moderated effect sizes for expressive language. As children with NSCL/P aged, their expressive language performance became more similar to their peers. Expressive language effect sizes from parent reports and observational language measures (estimated effect size = -0.74) were significantly lower than those from standardized norm-referenced tests (estimated effect size = -0.45). Conclusions These findings suggest that young children with NSCL/P experience delays relative to their peers across multiple speech and language constructs. Differences between children with NSCL/P and their typically developing peers appear to decrease with age. Supplemental Material https://doi.org/10.23641/asha.11356904.

Toward a paradigm shift from deficit-based to proactive speech and language treatment: Randomized pilot trial of the Babble Boot Camp in infants with classic galactosemia.

Background: Speech and language therapy is typically initiated reactively after a child shows delays. Infants with classic galactosemia (CG), a metabolic disease with a known high risk for both speech and language disorders, hold the keys towards evaluating whether preventive treatment is effective when the risks are known at birth. We present pilot data from a randomized parallel trial of an innovative proactive speech and language intervention program, the Babble Boot Camp (BBC).  Method: Five children with CG, otherwise healthy, participated in the study from approximately 2 to 24 months of age. One of these was randomly selected as control receiving conventional management, which typically starts at age 2-3 years. A pediatric speech-language pathologist met weekly via telepractice with the parents in the treatment cohort. Parents implemented the prespeech, speech, and language stimulation and expansion activities according to the intervention protocol. The control child was still too young for conventional treatment. Primary outcome measures were speech sound production complexity in babble and speech and expressive vocabulary size. Secondary outcome measures were vocalization rates and developmental milestones in communication, motor, and cognition. The trial is ongoing. Results:  All four treated children had higher speech sound skills in babble, three had higher speech sound skills in meaningful speech, two had higher expressive vocabularies, three had higher global developmental scores, and two had higher vocalization rates, compared to the control child with CG. Discussion: Given the high risk for speech and language delays in children with CG, finding on-schedule abilities in two or more of the treated children but not the untreated child is unexpected under random conditions. The trends toward beneficial effects of the BBC on speech sound production, expressive language, and communication milestones warrant appropriately powered larger clinical trials with full randomization. Trial registration: ClinicalTrials.gov NCT03838016 (12 th February 2019).