Speech in 7- and 10-year-olds born with a unilateral cleft lip and palate: a continued prospective Swedish intercentre study.

The aim of this study was to report longitudinal speech results in consecutively selected children from each of the six cleft centres in Sweden and to compare the results between centres. The children were born with a non-syndromic unilateral cleft lip and palate, and results from the same cohort at 5 years of age have previously been reported. Background data on medical care in terms of surgery, speech therapy, and hearing between 5 and 10 years of age were collected. Speech recordings of 56 children at 7 years and 54 at 10 years of age were blindly and independently assessed by four speech-language pathologists experienced in cleft palate speech. This resulted in measures of percent consonant correct (PCC) and perceived velopharyngeal competence rated on a three-tier scale. No statistically significant differences were found between centres. PCC scores at 7 years of age ranged from 44-100% (median 97.5) and at 10 years of age from 86-100% (median 100). Competent or marginally incompetent velopharyngeal function was found in 95% of the 7-year-olds and 98% of the 10-year-olds. Speech results were slightly better than previous reports of speech in children born with a unilateral cleft lip and palate.

Timing of Primary Surgery for Cleft Palate.

BACKGROUND: Among infants with isolated cleft palate, whether primary surgery at 6 months of age is more beneficial than surgery at 12 months of age with respect to speech outcomes, hearing outcomes, dentofacial development, and safety is unknown. METHODS: We randomly assigned infants with nonsyndromic isolated cleft palate, in a 1:1 ratio, to undergo standardized primary surgery at 6 months of age (6-month group) or at 12 months of age (12-month group) for closure of the cleft. Standardized assessments of quality-checked video and audio recordings at 1, 3, and 5 years of age were performed independently by speech and language therapists who were unaware of the trial-group assignments. The primary outcome was velopharyngeal insufficiency at 5 years of age, defined as a velopharyngeal composite summary score of at least 4 (scores range from 0 to 6, with higher scores indicating greater severity). Secondary outcomes included speech development, postoperative complications, hearing sensitivity, dentofacial development, and growth. RESULTS: We randomly assigned 558 infants at 23 centers across Europe and South America to undergo surgery at 6 months of age (281 infants) or at 12 months of age (277 infants). Speech recordings from 235 infants (83.6%) in the 6-month group and 226 (81.6%) in the 12-month group were analyzable. Insufficient velopharyngeal function at 5 years of age was observed in 21 of 235 infants (8.9%) in the 6-month group as compared with 34 of 226 (15.0%) in the 12-month group (risk ratio, 0.59; 95% confidence interval, 0.36 to 0.99; P = 0.04). Postoperative complications were infrequent and similar in the 6-month and 12-month groups. Four serious adverse events were reported (three in the 6-month group and one in the 12-month group) and had resolved at follow-up. CONCLUSIONS: Medically fit infants who underwent primary surgery for isolated cleft palate in adequately resourced settings at 6 months of age were less likely to have velopharyngeal insufficiency at the age of 5 years than those who had surgery at 12 months of age. (Funded by the National Institute of Dental and Craniofacial Research; TOPS ClinicalTrials.gov number, NCT00993551.).

Inter-rater reliability in classification of canonical babbling status based on canonical babbling ratio in infants with isolated cleft palate randomised to Timing of Primary Surgery for Cleft Palate (TOPS).

Canonical babbling (CB) is commonly defined as present when at least 15% of all syllables produced are canonical, in other words a canonical babbling ratio (CBR) ≥0.15. However, there is limited knowledge about inter-rater reliability in classification of CB status based on CBR and inter-rater differences in assessment of CBR. We investigated inter-rater reliability of experienced Speech Language Therapists (SLTs) on: classification of CB status based on CBR ≥ 0.15, CBRs and the total number of syllables per infant used to calculate CBR.Each infant (n = 484) was video-recorded at a clinical site in play interaction with their parent as part of the randomised controlled trial Timing of Primary Surgery for Cleft Palate. Each recording was subsequently assessed by three independent SLTs, from a pool of 29 SLTs. They assessed the recordings in real time.The three assessing SLTs agreed in classification of CB status in 423 (87.4%) infants, with higher complete agreement for canonical (91%; 326/358) than non-canonical (77%; 97/126). The average difference in CBR and total number of syllables identified between the SLT assessments of each infant was 0.12 and 95, respectively.This study provided new evidence that one trained SLT can reliably classify CB status (CBR ≥ 0.15) in real time when there is clear distinction between the observed CBR and the boundary (0.15); however, when the observed CBR approaches the boundary multiple SLT assessments are beneficial. Thus, we recommend to include assessment of inter-rater reliability, if the purpose is to compare CBR and total syllable count across infants or studies.Trial registration number here: www.clinicaltrials.gov, identifier NCT00993551.

Reliability of data on percent consonants correct and its associated quality indicator in the Swedish cleft lip and palate registry.

BACKGROUND: Data in national health care quality registries must be valid and reliable in order to enable open comparisons of results. AIM: To assess the reliability of data on percent consonants correct (PCC) and its associated quality indicator ≥86% correct consonants in the Swedish quality registry for patients born with cleft lip and palate (CLP) registry. METHODS: Six independent speech-language pathologists re-assessed the audio recordings of 96 five-year-olds with PCC data in the CLP registry. Target consonants of a single-word picture-naming test were phonetically transcribed, and PCC was calculated. The reliability of PCC data was assessed with the intraclass correlation coefficient (ICC). The reliability of the quality indicator ≥86% correct consonants was assessed with point-by-point percentage agreement and Cohen's kappa. RESULTS: Intra- and inter-judge agreement for PCC was excellent with ICCs above 0.9, and so was the agreement of data from the CLP registry and the six judges' re-assessments. The percentage agreement between all judges and the CLP registry for the quality indicator ≥86% correct consonants was poor (67%). However, in 88% of the cases, results from four judges and the CLP registry agreed, corresponding to good agreement. The mean of all kappa values for six judges and the CLP registry corresponded to good agreement (0.72). CONCLUSIONS: The results indicate the PCC data in the CLP registry and the quality indicator ≥86% correct consonants to be reliable. When differences in outcome between treatment centres are detected, the raw data collected should always be re-examined before drawing definitive conclusions.

Inter-centre comparison of data on surgery and speech outcomes at 5 years of age based on the Swedish quality registry for patients born with cleft palate with or without cleft lip.

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.

Coverage, reporting degree and design of the Swedish quality registry for patients born with cleft lip and/or palate.

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.

Reliability of Speech Variables and Speech-Related Quality Indicators in the Swedish Cleft Lip and Palate Registry.

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.

Speech in 5-year-olds born with unilateral cleft lip and palate: a Prospective Swedish Intercenter Study.

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.

Comparison between perceptual assessments of nasality and nasalance scores.

BACKGROUND: There are different reports of the usefulness of the Nasometer™ as a complement to listening, often as correlation calculations between listening and nasalance measurements. Differences between findings have been attributed to listener experience and types of speech stimuli. AIMS: To compare nasalance scores from the Nasometer with perceptual assessments, for the same and different Swedish speech stimuli, using three groups of listeners with differing levels of experience in judging speech nasality. METHODS & PROCEDURES: To compare nasalance scores and blinded listener ratings of randomized recordings using three groups of listeners and two groups of speakers. Speakers were either classified as having hypernasal speech or speech with typical speech resonance. Listeners were speech-language pathologists (SLPs) working predominantly with resonance disorders, other SLPs and untrained listeners. OUTCOMES & RESULTS: Correlations (r(s)) between hypernasality ratings and nasalance scores for each listener group and speech stimuli were calculated. For both groups of SLPs all correlations between perceptual ratings and nasalance scores were significant at p= 0.01. The correlations between the nasalance scores and ratings by listeners in the SLP groups were higher than those for the untrained listener group regardless of stimulus type. Post-hoc Mann-Whitney U-tests showed that the only difference that was significant was expert SLP group versus untrained listener group. Secondly, correlations between perceptual ratings and oral stimulus nasalance scores were higher when the perceptual ratings were based on spontaneous speech rather than on the oral stimulus. However, a Wilcoxon signed rank test showed that the difference was not significant. A third finding was that correlations between oral stimulus nasalance scores and perceptual scores were higher than those between mixed stimulus nasalance scores and perceptual scores. A Wilcoxon signed rank test showed that the difference was significant. CONCLUSIONS & IMPLICATIONS: The Nasometer might be useful for the SLP with limited experience in assessing resonance disorders in differentiating between hyper- and hyponasality. With listener reliability for ratings of hypernasality still being an issue, the use of a nasalance score as a complement to the perceptual evaluation will also aid the expert SLP. It will give an alternative way of quantifying speech resonance and might help in especially hard to judge cases.

Normative data on nasalance scores for Swedish as measured on the Nasometer: influence of dialect, gender, and age.

This study was conducted to establish normative nasalance values for Swedish speaking children as measured with the Nasometer(trade mark) II, and to investigate differences due to regional dialect, gender, and age. Two hundred and twenty healthy children aged 4-5, 6-7, and 9-11 years were included. Group mean nasalance scores for four speech stimuli were calculated and compared. There were no significant differences due to dialect or gender for children in the study. For age there was a significant difference on nasal sentences between the youngest group of children and the other two groups, age 4-5 vs age 6-7 (t = -2.844, p = .006) and for age 4-5 vs age 9-11 (t = -2.888, p = .005). The results from this study have both clinical significance for Swedish SLPs working with resonance disorders, and theoretical significance for linguists studying features of dialects and languages.

Untrained listeners' ratings of speech disorders in a group with cleft palate: a comparison with speech and language pathologists' ratings.

BACKGROUND: Hypernasal resonance, audible nasal air emission and/or nasal turbulence, and articulation errors are typical speech disorders associated with the speech of children with cleft lip and palate. Several studies indicate that hypernasal resonance tends to be perceived negatively by listeners. Most perceptual studies of speech disorders related to cleft palate are carried out with speech and language pathologists as listeners, whereas only a few studies have been conducted to explore how judgements by untrained listeners compare with expert assessments. These types of studies can be used to determine whether children for whom speech and language pathologists recommend intervention have a significant speech deviance that is also detected by untrained listeners. AIMS: To compare ratings by untrained listeners with ratings by speech and language pathologists for cleft palate speech. METHODS & PROCEDURES: An assessment form for untrained listeners was developed using statements and a five-point scale. The assessment form was tailored to facilitate comparison with expert judgements. Twenty-eight untrained listeners assessed the speech of 26 speakers with cleft palate and ten speakers without cleft in a comparison group. This assessment was compared with the joint assessment of two expert speech and language pathologists. OUTCOMES & RESULTS: Listener groups generally agreed on which speakers were nasal. The untrained listeners detected hyper- and hyponasality when it was present in speech and considered moderate to severe hypernasality to be serious enough to call for intervention. The expert listeners assessed audible nasal air emission and/or nasal turbulence to be present in twice as many speakers as the untrained listeners who were much less sensitive to audible nasal air emission and/or nasal turbulence. CONCLUSIONS & IMPLICATIONS: The results of untrained listeners' ratings in this study in the main confirm the ratings of speech and language pathologists and show that cleft palate speech disorders may have an impact in the everyday life of the speaker.

Optimizing stimulus length for clinical nasalance measures in Swedish.

Standardized passages used for speech nasalance measures may be too long for clinical use with very young or non-compliant patients. The aim of this study was to establish whether nasalance scores from shorter sections of three Swedish speech stimuli were equivalent to those from their corresponding whole stimulus. Nasalance recordings for three Swedish speech stimuli (oral, nasal and oronasal) were obtained from 29 typically developing Swedish children (7-11 years). Cumulative sentence combinations were evaluated for equivalence to their respective whole passages according to two different criteria: one based on t-testing and the other on cumulative frequencies of score differences. Results showed that shorter sequences of sentences could be considered equivalent to the whole passage for the oral and nasal stimuli, provided that the sentences were sequenced in order of increasing difference from the whole stimulus score and that those sentences with scores significantly different from the whole passage score were not included.

A cross-sectional study of speech in 10-year-old children with cleft palate: results and issues of rater reliability.

OBJECTIVES: To describe speech based on perceptual evaluation in a group of 10-year-old children with cleft palate. A secondary aim was to investigate the reliability of speech-language pathologists' perceptual assessment of cleft palate speech. DESIGN: Retrospective cross-sectional study in children with cleft palate. External raters made assessments from randomized speech recordings. SUBJECTS: Thirty-eight children with unilateral cleft lip and palate (UCLP) or cleft palate only (CPO) and 10 children in a comparison group. MAIN OUTCOME MEASURES: Ratings of hypernasality, hyponasality, audible nasal air leakage, weak pressure consonants, and articulation. Exact agreement and weighted kappa values were used for reliability. RESULTS: Hypernasality was found in 25% of children with a cleft of the soft palate (CSP), 33% of children with a cleft of the hard and soft palate (CHSP), and 67% of children with a UCLP. Similar results were found for audible nasal air leakage. Articulation errors were found in 6% of the CHSP group and 25% of the UCLP group, whereas no child in the CSP group had articulation errors. The reliability was moderate to good for different variables, with lowest values for hypernasality. CONCLUSIONS: Speech results in this series seem less satisfactory than those reported in other published international studies, but it is difficult to draw any certain conclusions about speech results because of large methodological differences. Further developments to ensure high reliability of perceptual ratings of speech are called for.