Overall weighted or composite variables for perceptual auditory estimation of velopharyngeal closure or competence have been used in several studies for evaluation of velopharyngeal function during speech. The aim of the present study was to investigate the validity of a composite score (VPC-Sum) and of auditory perceptual ratings of velopharyngeal competence (VPC-Rate). Available VPC-Sum scores and judgments of associated variables (hypernasality, audible nasal air leakage, weak pressure consonants, and non-oral articulation) from 391 5-year olds with repaired cleft palate (the Scandcleft project) were used to investigate content validity, and 339 of these were compared with an overall judgment of velopharyngeal competence (VPC-Rate) on the same patients by the same listeners. Significant positive correlations were found between the VPC-Sum and each of the associated variables (Cronbachs alpha 0.55-0.87, P < 0.001), and a moderately significant positive correlation between VPC-Sum and VPC-Rate (Rho 0.698, P < 0.01). The latter classified cases well when VPC-Sum was dichotomized with 67% predicted velopharyngeal competence and 90% velopharyngeal incompetence. The validity of the VPC-Sum was good and the VPC-Rate a good predictor, suggesting possible use of both measures depending on the objective.
Maxillary advancement performed to normalize occlusion and facial profile improved consonant proficiency in patients with CLP 1 year postoperation. Lay listeners' and patients' perceptions of speech need further exploration.
Purpose: Cleft lip and/or palate (CLP) is a common birth defect, and after reconstructive surgery, about 50% of children at 5 years of age have speech deviations and are referred to speech-language therapy (SLT). The peer-reviewed evidence for the benefit of SLT has been uncertain. Our objective was to systematically review and meta-analytically summarize the benefit of SLT for individuals born with CLP. Method: A systematic search was conducted (last search on February 19, 2021) on studies evaluating SLT with pre and post measures on speech production, language ability, intelligibility, and/or patient-reported outcomes. We sought individual participant data (IPD) and evaluated on an individual level if the outcome measure had improved to a clinically relevant degree during SLT and if the outcome measure was on a level with peers or not after SLT. Meta-analyses and meta-regressions were applied to synthesize IPD across studies. Results: Thirty-four eligible studies were found. Nineteen studies provided IPD ( n = 343) for the main analysis on speech production. The synthesized information suggests that, during SLT, speech production improved to a clinically relevant degree for many individuals (95% CI [61%, 87%]) and that speech production was on a level with peers for some individuals after SLT (95% CI [10%, 34%]). Conclusions: The main strength of this meta-analysis is that we evaluated on an individual level pre- and post-intervention data based on considerations of clinical relevance. This approach allowed us to conclude that many individuals benefit from SLT and that further work on evaluating SLT in this patient group is meaningful. Supplemental Material https://doi.org/10.23641/asha.17700992
Objective: To assess the reliability of speech variables and speech-related quality indicators in the Swedish quality registry for cleft lip and palate (CLP). Design: Retrospective study. Setting: Primary care university hospitals. Participants: Fifty-two 5-year-old children with unilateral CLP and 41 with bilateral CLP. Main Outcome Measures: Registry data for “percent nonoral errors” and “perceived velopharyngeal competence” (VPC) were compared to reassessments by 4 independent judges based on audio recordings. Interjudge agreement for “percent consonants correct” (PCC) and the reliability of 3 quality indicators were also assessed. Agreement was calculated with single measures intraclass correlation coefficient (ICC) for articulation outcomes, quadratic weighted κ and ICC for VPC, and percentage agreement and κ for quality indicators. Results: When the agreement between registry data and the judges’ reassessments was assessed, the ICC was 0.79 for percent nonoral errors. For VPC, the κ coefficient was 0.66 to 0.75 and the ICC was 0.73. Interjudge agreement for PCC calculated with ICC was 0.85. For the quality indicator “proportion of children with ≥86% correct consonants,” all 4 judges were in agreement for 72% of the cases. For “proportion of children without nonoral speech errors” and “proportion of children with competent or marginally incompetent velopharyngeal function,” the agreement between registry data and the 4 judges was 89% and 85%, respectively. Conclusions: The results indicate that registry data on PCC, percent nonoral errors, VPC, and the quality indicators “proportion of children without nonoral speech errors” and “proportion of children with competent or marginally incompetent velopharyngeal function” are reliable.
Background: The objective of the Swedish cleft lip and palate (CLP) registry is to promote quality control, research and improvement of treatment, by comparison of the long-term results of surgery, orthodontics and speech from all six Swedish CLP centres. The purpose of the study was to investigate the coverage and reporting degree of the Swedish CLP registry, and to describe the design of the registry and discuss questions of reliability and validity of the data included. Methods: All six Swedish CLP centres participate in the registry. All children in Sweden with cleft lip and/or cleft palate, born from 2009 onwards, are included in the registry. Baseline data such as cleft type (ICD-10 diagnosis), heredity, birth weight and additional deformities and/or syndromes, as well as pre-surgical treatment, are recorded at first visit. Data on surgical treatment are recorded continuously. Treatment outcome regarding dentofacial development and speech are recorded at follow-ups at 5, 10, 16 and 19 years of age. Data on dentofacial development are also recorded 1 year after orthognathic surgery. In addition, data on babbling and speech are recorded at 18 months of age. Coverage degree and reporting degree of surgery was assessed by comparison with registrations in the Swedish Central patient registry. Reporting degree of orthodontic and speech registrations at 5 years of age was assessed by comparison with registrations at baseline. Results: The average coverage degree for children born 2009 to 2018 was 95.1%. For cleft-related surgeries, the average reporting degree was 92.4%. Average reporting degree of orthodontic registrations and speech registrations at age 5 years was 92 and 97.5% respectively. Conclusion: In order to achieve valid and reliable data in a healthcare quality registry, the degree of coverage and reporting needs to be high, the variables included should be limited and checked for reliability, and the professionals must calibrate themselves regularly. The Swedish CLP registry fulfils these requirements.
Objective: The aim of this study was to explore how 7-year-olds describe speech in children born with cleft palate in their own words and to investigate whether they perceive signs of velopharyngeal incompetence (VPI) and articulation errors, and if so, which terminology they use. Methods/Participants: Twenty 7-year-olds participated in 6 focus group interviews where they listened to 8 speech samples with different types of cleft speech characteristics and described what they heard. The same speech samples had been assessed by speech-language pathologists and comprised normal speech, different degrees of VPI, oral articulation disorders, and glottal articulation. The interviews were analyzed with qualitative content analysis. Results: The analysis resulted in 4 interlinked categories: descriptions of speech, thoughts on personal traits, consequences for communication, and emotional reactions and associations. Each category contains 4 to 5 subcategories with the children’s descriptions and reflections. Glottal articulation and severe signs of VPI caused the most negative emotional reactions and were described as sounding scary and incomprehensible and the children speculated on the risk of social rejection of the speakers. Retracted oral articulation was also noted and described but with a vocabulary similar to the professionals. Minor signs of VPI were not noted. Conclusions: Seven-year-olds are direct and straightforward in their reactions to cleft palate speech characteristics. More pronounced signs of VPI and articulatory difficulties, also minor ones, are noted. Clinically, articulatory impairments may be more important to treat than minor signs of VPI.
Studies on the impact of cleft palate surgery on speech with stringent methodology are called for, since we still do not know the best timing or the best method for surgery. The purpose was to report on speech outcome for all Swedish-speaking 5-year-olds born with a non-syndromic unilateral cleft lip and palate (UCLP), in 2008-2010, treated at Sweden's six cleft palate centres, and to compare speech outcomes between centres. Speech was assessed in 57 children with percent consonants correct adjusted for age (PCC-A), based on phonetic transcriptions from audio recordings by five independent judges. Also, hypernasality and perceived velopharyngeal function were assessed. The median PCC-A for all children was 93.9, and medians in the different groups varied from 89.9 to 96.8. In the total group, 9 children (16%) had more than mild hypernasality. Twenty-two children (38.5%) were perceived as having competent/sufficient velopharyngeal function, 25 (44%) as having marginally incompetent/insufficient velopharyngeal function, and 10 children (17.5%) as having incompetent/insufficient velopharyngeal function. Ten children were treated with secondary speech improving surgery and/or fistula surgery. No significant differences among the six groups, with eight to ten children in each group, were found. The results were similar to those in other studies on speech of children with UCLP, but poorer than results in normative data of Swedish-speaking 5-year-olds without UCLP. Indications of differences in frequency of surgical treatment and speech treatment between centres were observed.
Background The objective of the Swedish cleft lip and palate registry (CLP registry) is to promote quality control, research and improvement of treatment, by the comparison of long-term results. The aim was to compare data from the CLP registry among the six treatment centres, regarding data on surgery and speech outcomes at 5 years of age. Methods The participants were 430 children born in Sweden from 2009 to 2014, with cleft palate with or without cleft lip and without known syndromes and/or additional malformations. The number of primary and secondary palatal surgeries up to 5 years of age, timing of the last primary palatal surgery, percentage consonants correct, percentage non-oral speech errors and perceived velopharyngeal competence at 5 years were assessed. Multivariable binary logistic regression adjusted for sex and cleft type was used to compare results between the six centres. Results At one centre (centre 4), the palate was closed in one to three stages, and at the remaining centres in one or two stages. At centre 4, more children underwent a higher number of palatal surgeries, and the last primary palatal surgery was performed at a higher age. Children in centre 4 were also less likely to achieve ≥86% correct consonants (OR = 0.169, P = < 0.001), have no non-oral speech errors (OR = 0.347, P = < 0.001), or have competent or marginally incompetent velopharyngeal competence (OR = 0.244, P = < 0.001), compared to the average results of the other centres. No clear association between patient volume and speech outcome was observed. Conclusions The results indicated the risk of a negative speech result if the last primary palatal surgery was performed after 25 months of age. Whether the cleft in the palate was closed in one or two stages did not affect speech outcome. The Swedish CLP registry can be used for open comparisons of treatment results to provide the basis for improvements of treatment methods. If deviating negative results are seen consistently at one centre, this information should be acted upon by further investigation and analysis, making changes to the treatment protocol as needed.
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