Objective
To assess the ability of a cleft-specific multi-site learning health network registry to describe variations in cleft outcomes by cleft phenotypes, ages, and treatment centers. Observed ...variations were assessed for coherence with prior study findings.
Design
Cross-sectional analysis of prospectively collected data from 2019–2022.
Setting
Six cleft treatment centers collected data systematically during routine clinic appointments according to a standardized protocol.
Participants
714 English-speaking children and adolescents with non-syndromic cleft lip/palate.
Intervention
Routine multidisciplinary care and systematic outcomes measurement by cleft teams.
Outcome Measures
Speech outcomes included articulatory accuracy measured by Percent Consonants Correct (PCC), velopharyngeal function measured by Velopharyngeal Competence (VPC) Rating Scale (VPC-R), intelligibility measured by caregiver-reported Intelligibility in Context Scale (ICS), and two CLEFT-Q™ surveys, in which patients rate their own speech function and level of speech distress.
Results
12year-olds exhibited high median PCC scores (91–100%), high frequency of velopharyngeal competency (62.50–100%), and high median Speech Function (80–91) relative to younger peers parsed by phenotype. Patients with bilateral cleft lip, alveolus, and palate reported low PCC scores (51–91%) relative to peers at some ages and low frequency of velopharyngeal competency (26.67%) at 5 years. ICS scores ranged from 3.93–5.0 for all ages and phenotypes. Speech Function and Speech Distress were similar across phenotypes.
Conclusions
This exploration of speech outcomes demonstrates the current ability of the cleft-specific registry to support cleft research efforts as a source of “real-world” data. Further work is focused on developing robust methodology for hypothesis-driven research and causal inference.
Aim
This study evaluated early speech and language development at 18 and 24 months, and associated factors, based on parental reports.
Method
We followed up the CHILD‐SLEEP birth cohort of 1667 ...Finnish‐speaking families, who were randomly recruited in 2011–2013 during routine visits to maternity clinics in the Pirkanmaa Hospital District of Finland. The women were approximately 32 weeks’ pregnant at enrolment. Parents reported the size of their child's expressive vocabulary, word combinations, intelligibility, finger‐pointing and adherence to instructions. A subsample was studied using the Expressive Language subscale of the Bayley Scales of Infant and Toddler Development, Third Edition.
Results
The children's vocabulary was smaller than previously reported. At 18 months of age, 68.8% of the 997 children had a vocabulary of 20 words or less and 35.7% used about five words at most. At 24 months, 32.4% of the 822 children had a vocabulary of 50 words or less and 18.4% used about 20 words at most. Longer child and parental exposure to electronic media was negatively associated with the size of the child's expressive vocabulary.
Conclusion
Vocabulary size at 18 and 24 months was smaller than previously reported and negatively associated with exposure to electronic media.
Speech disfluencies such as repeated words and pauses provide information about the cognitive systems underlying speech production. Understanding whether older age leads to changes in speech fluency ...can therefore help characterize the robustness of these systems over the life span. Older adults have been assumed to be more disfluent, but current evidence is minimal and contradictory. Particularly noteworthy is the lack of longitudinal data that would help establish whether a given individual's disfluency rates change over time. This study examines changes in disfluency rates through a sequential design with a longitudinal component, involving the analysis of 325 recorded interviews conducted with 91 individuals at several points in their lives, spanning the ages of 20-94 years. We analyzed the speech of these individuals to assess the extent to which they became more disfluent in later interviews. We found that, with older age, individuals spoke more slowly and repeated more words. However, older age was not associated with other types of disfluencies such as filled pauses (uh's and um's) and repairs. Overall, this study provides evidence that, although age itself is not a strong predictor of disfluencies, age leads to changes in other speech characteristics among some individuals (i.e., speech rate and indicators of lexical and syntactic complexity), and those changes in turn predict the production of disfluencies over the life span. These findings help resolve previous inconsistencies in this literature and set the stage for future experimental work on the cognitive mechanisms underlying changes in speech production in healthy aging.
Public Significance Statement
This study measures changes in speech fluency over the life span sequentially using a novel corpus of 91 individuals interviewed at multiple times in their lives. We found that, while older adults spoke more slowly and repeated more words, they were not more likely to produce filled pauses (uh's and um's) and repairs. Overall, we suggest that age is not as strong a predictor of disfluencies as are other individual differences in speech characteristics.
The purpose of this study was to investigate the cross-linguistic influences of Jamaican Creole (JC) and English on phonemic and vocal development in bilingual JC-English-speaking preschoolers.
...Sixteen typically developing children (12 females,
= 4 years; 4 months) completed the Diagnostic Evaluation of Articulation in Phonology Word Inconsistency Assessment subtest in JC and in English. Acoustic measures of voice onset time (VOT), VOT variability (VOT
), mean fundamental frequency (
), and
variability (
) were extracted from each target word. Prevoicing was noted. Mixed models and regression models were analysed to understand the patterns of acoustic measures in each language, and the relationship between phonemic and vocal variability, respectively.
Analyses showed a significant effect of language on
, wherein
was greater in English than JC. JC spoken (percentage) was a significant positive predictor of VOT
for voiced (short lag) productions. There was no relationship between phonemic and vocal variability measures.
Greater
in English may be due to linguistic
differences and speaking environment. Variability for voiced VOT is likely due to the continued maturation of vocal and articulatory control when children are developing adult-like productions, though longitudinal studies are needed.
Objective
Measuring disability as a concept of impaired global function enables beneficiaries of treatment, the impact of treatment, and targets of health system investment to be rigorously assessed. ...Measures of disability are not well established for cleft lip and palate. This study aims to systematically review disability weight (DW) studies pertaining to orofacial clefts (OFCs) and identify methodological strengths and shortcomings of each approach.
Design
Systematic literature review of studies that met the following criteria: (1) peer-reviewed publication, (2) focus on disability valuation, (3) mention orofacial clefts, and (4) publication January 2001-December 2021.
Setting
None.
Patients/Participants
None.
Interventions
None.
Main Outcome Measure(s)
Disability weight method of valuation and the value itself.
Results
The final search strategy yielded 1,067 studies. Seven manuscripts were ultimately included for data extraction. The disability weights used in our studies, including those newly generated or taken from the Global Burden of Disease Studies (GBD), ranged widely for isolated cleft lip (0.0–0.100) and cleft palate with or without cleft lip (0.0−0.269). The GBD studies limited their consideration of cleft sequelae informing disability weights to impact on appearance and speech-related concerns, while other studies accounted for comorbidities such as pain and social stigma.
Conclusions
Current measures of cleft disability are sparse, inadequately reflect the comprehensive impact of an OFC on function and socialization, and are limited in detail or supporting evidence. Use of a comprehensive health state description in evaluating disability weights offers a realistic means of accurately representing the diverse sequelae of an OFC.
Objective
To compare the effectiveness of short-term ventilation tubes compared to surveillance on conductive hearing loss in children with non-syndromic orofacial clefting involving the muscular ...palate.
Introduction
Chronic otitis media with effusion is a common finding in children with cleft palate. The accepted convention is insertion of short-term ventilation tubes at the time of palate repair, but some centres are choosing conservative management. Each approach has its advantages but there is currently no consensus on the most appropriate management in children with non-syndromic cleft palate.
Inclusion criteria
Children <18 years with cleft lip and palate, or isolated cleft palate, not associated with a genetic syndrome, who have been diagnosed with chronic otitis media with effusion.
Methods
A systematic search of MEDLINE, CINAHL, Embase and Scopus databases was conducted. Grey literature searches were conducted through Central Register of Controlled Trials, Clinicaltrials.gov and ProQuest. Two reviewers screened the studies, conducted critical appraisal, assessed the methodological quality, and extracted the data. Where possible, studies were pooled in statistical meta-analysis with heterogeneity being assessed using the standard Chi-squared and I2 tests.
Results
Four studies met the inclusion criteria but were of low quality with a moderate risk of bias. Only data on hearing thresholds could be pooled for analysis which found no statistically significant difference. Other outcomes were presented in narrative form. Certainty of evidence for all outcomes was deemed low to very low using GRADE criteria.
Conclusions
No definitive conclusions can be drawn regarding most effective management at improving conductive hearing loss. Missing data and inconsistent reporting of outcomes limited capacity for pooled analysis.
Objective
Lambdoid craniosynostosis affects approximately 1 in 33 000 live births per year, and surgical correction is often sought in order to achieve normocephaly and allow for adequate brain ...growth. However, the effects of lambdoid synostosis and its treatment on cognitive development are unknown.
Design
Systematic review and meta-analysis.
Patients, Participants
A systematic review of Ovid Medline, Embase, Web of Science, Scopus, Cochrane Central Register of Controlled Trials, and Clinicaltrials.gov was conducted in January 2022. Included studies assessed cognitive development of patients with nonsyndromic unilateral lambdoid craniosynostosis.
Main Outcome Measures
Results of developmental tests were compared to normative data or controls to generate Hedges’ g for meta-analysis.
Results
Nine studies met the inclusion criteria. Meta-analysis of 3 studies describing general cognition showed that cases scored significantly lower than their peers, but within 1 standard deviation (g = 0.37, 95% CI −0.64, −0.10, P = .01). Meta-analysis of verbal and psychomotor development showed no significant differences in children with lambdoid synostosis. Studies were of fair quality and had moderate-to-high heterogeneity.
Conclusions
Patients with lambdoid craniosynostosis may score slightly below average on tests of general cognition in comparison to normal controls, but results in other domains are variable. Analyses were limited by small sample sizes. Multidisciplinary care and involvement of a child psychologist may be helpful in identifying areas of concern and providing adequate scholastic support. Further research recruiting larger cohorts will be necessary to confirm these findings and extend them to other developmental domains such as attention and executive function.
The present study investigates the relation between language environment and language delay in 63 British-English speaking children (19 typical talkers (TT), 22 late talkers (LT), and 22 late ...bloomers (LB) aged 13 to 18 months. Families audio recorded daily routines and marked the new words their child produced over a period of 6 months. To investigate how language environments differed between talker types and how environments corresponded with children's developing lexicons, we evaluated contextual diversity-a word property that measures semantic richness-and network properties of language environments in tandem with developing vocabularies. The language environment experienced by the three talker types differed in their structural properties, with LT environments being least contextually diverse and least well-connected in relation to network properties. Notably, LBs' language environments were more like those of TTs. Network properties of language environments also correlate with the rate of vocabulary growth over the study period. By comparing differences between language environments and lexical network development, we also observe results consistent with contributions to lexical development from different learning strategies for expressive vocabularies and different environments for receptive vocabularies. We discuss the potential consequences that structural differences in parental speech might have on language development and the contribution of this work to the debate on quantity versus quality.
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