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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.).
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Fissura Palatina , Insuficiência Velofaríngea , Pré-Escolar , Humanos , Lactente , Pessoal Técnico de Saúde , Fissura Palatina/complicações , Fissura Palatina/cirurgia , Europa (Continente) , Complicações Pós-Operatórias/epidemiologia , Insuficiência Velofaríngea/diagnóstico , Insuficiência Velofaríngea/etiologia , América do Sul , Técnicas de Diagnóstico por CirurgiaRESUMO
AIM: Speech difficulties are common in children with cleft palate, but research on foreign-born children is limited. This study aimed to compare speech outcomes, surgery and speech intervention in 5-year-old foreign-born and Swedish-born children with cleft palate with or without cleft lip. METHODS: This retrospective study analysed data from the Swedish cleft lip and palate registry for children born between 2009 and 2016 using Pearson's Chi-squared test and binary logistic regression. RESULTS: Among 160 foreign-born (106 boys, 54 girls) and 847 Swedish-born (479 boys, 368 girls) 5-year-olds, foreign-born children had significantly lower rates of sufficient velopharyngeal competence (77% vs. 86%), age-appropriate consonant production (28% vs. 60%), and speech without non-oral speech errors (70% vs. 86%). Differences remained after adjustment for cleft type, gender and additional diagnosed conditions. After further adjustments for age at completed primary palatal surgery, differences in age-appropriate consonant production and speech without non-oral speech errors remained significant. Foreign-born children underwent completed primary palatal surgery at older ages and received more secondary palatal surgery and speech intervention than Swedish-born peers. CONCLUSION: Foreign-born children showed poorer speech outcomes than Swedish-born peers, despite more secondary palatal surgery and speech intervention. Age at completed primary palatal surgery could partly explain these differences.
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OBJECTIVE: Development of an AI tool to assess velopharyngeal competence (VPC) in children with cleft palate, with/without cleft lip. DESIGN: Innovation of an AI tool using retrospective audio recordings and assessments of VPC. SETTING: Two datasets were used. The first, named the SR dataset, included data from follow-up visits to Skåne University Hospital, Sweden. The second, named the SC + IC dataset, was a combined dataset (SC + IC dataset) with data from the Scandcleft randomized trials across five countries and an intercenter study performed at six Swedish CL/P centers. PARTICIPANTS: SR dataset included 153 recordings from 162 children, and SC + IC dataset included 308 recordings from 399 children. All recordings were from ages 5 or 10, with corresponding VPC assessments. INTERVENTIONS: Development of two networks, a convolutional neural network (CNN) and a pre-trained CNN (VGGish). After initial testing using the SR dataset, the networks were re-tested using the SC + IC dataset and modified to improve performance. MAIN OUTCOME MEASURES: Accuracy of the networks' VPC scores, with speech and language pathologists scores seen as the true values. A three-point scale was used for VPC assessments. RESULTS: VGGish outperformed CNN, achieving 57.1% accuracy compared to 39.8%. Minor adjustments in data pre-processing and network characteristics improved accuracies. CONCLUSIONS: Network accuracies were too low for the networks to be useful alternatives for VPC assessment in clinical practice. Suggestions for future research with regards to study design and dataset optimization were discussed.
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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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Fenda Labial , Fissura Palatina , Criança , Pré-Escolar , Fenda Labial/complicações , Fenda Labial/cirurgia , Fissura Palatina/complicações , Fissura Palatina/cirurgia , Humanos , Sistema de Registros , Fala , Suécia , Resultado do TratamentoRESUMO
BACKGROUND: When evaluating speech in children with cleft palate with or without cleft lip (CP/L), children with known syndromes and/or additional malformations (CP/L+) are usually excluded. The aim of this study was to present speech outcome of a consecutive series of 5-year-olds born with CP/L, and to compare speech results of children with CP/L + and children with CP/L without known syndromes and/or additional malformations (CP/L-). METHODS: One hundred 5-year-olds (20 with CP/L+; 80 with CP/L-) participated. All children were treated with primary palatal surgery in one stage with the same procedure for muscle reconstruction. Three independent judges performed phonetic transcriptions and rated perceived velopharyngeal competence from audio recordings. Based on phonetic transcriptions, percent consonants correct (PCC) and percent non-oral errors were investigated. Group comparisons were performed. RESULTS: In the total group, mean PCC was 88.2 and mean percent non-oral errors 1.5. The group with bilateral cleft lip and palate (BCLP) had poorer results on both measures compared to groups with other cleft types. The average results of PCC and percent non-oral errors in the CP/L + group indicated somewhat poorer speech, but no significant differences were observed. In the CP/L + group, 25 % were judged as having incompetent velopharyngeal competence, compared to 15 % in the CP/L- group. CONCLUSIONS: The results indicated relatively good speech compared to speech of children with CP/L in previous studies. Speech was poorer in many children with more extensive clefts. No significant differences in speech outcomes were observed between CP/L + and CP/L- groups.
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Fenda Labial , Fissura Palatina , Criança , Pré-Escolar , Fenda Labial/complicações , Fenda Labial/cirurgia , Fissura Palatina/complicações , Fissura Palatina/cirurgia , Humanos , Fala , SíndromeRESUMO
AIM: The aim of this study was to investigate language ability in internationally adopted children aged 7-8 years with and without a unilateral cleft lip and palate. METHODS: We compared 27 internationally adopted children with a unilateral cleft lip and palate, adopted from China, with a group of 29 children without a cleft lip and palate, adopted from different countries. Participants were recruited from two cleft lip and palate teams in Sweden and through adoption organisations. Assessments were performed using standardised tests of speech and of receptive and expressive language ability. In addition, a parental questionnaire in which speech, language and communication aspects were rated was used. RESULTS: There were no significant differences in language ability between the groups. The only difference was related to speech ability, where the internationally adopted children with unilateral cleft lip and palate scored significantly lower. However, a high proportion of children in both groups scored low on measures of expressive language compared with test norms. CONCLUSION: The results suggested that having a cleft lip and palate did not increase the risk of language difficulties. Instead, being internationally adopted may be associated with a risk of delayed language development lasting for several years post-adoption.
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Criança Adotada , Fenda Labial , Fissura Palatina , Criança , China , Humanos , Instituições Acadêmicas , SuéciaRESUMO
OBJECTIVE: The primary aim of this study was to investigate whether there was any difference in scores of the Cleft Hearing, Appearance and Speech Questionnaire (CHASQ) between patients with cleft lip and/or cleft palate (CL/P) and a control population. The second aim was to compare CL/P and control population scores in this study with a British norm CL/P population. DESIGN: Single-site, cross-sectional study with an age-matched control population. SETTING: Participants were recruited from a hospital, a school, and a sports club. They answered the CHASQ in the hospital or at home. PARTICIPANTS: Sixty-four participants with CL/P (7-19 years of age) and a control population of 56 participants without CL/P (9-20 years of age). MAIN OUTCOME MEASURE: CHASQ. RESULTS: There was no statistically significant difference in satisfaction with cleft-related features between the CL/P and the control population. Participants with CL/P were significantly more satisfied with non-cleft-related features than the control population. Cleft Hearing, Appearance and Speech Questionnaire scores were also similar to earlier established British normative data of a CL/P population. CONCLUSION: The results indicated that children and young people with CL/P were as satisfied with their appearance, hearing, and speech as children and young people without CL/P. Swedish CHASQ scores were also similar to British scores.
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Fenda Labial , Fissura Palatina , Adolescente , Criança , Estudos Transversais , Audição , Humanos , Fala , Inquéritos e Questionários , SuéciaRESUMO
OBJECTIVE: The primary aim of this study was to compare corresponding scores between 2 existing cleft-specific patient-reported outcome measures (PROMs)-Cleft Hearing Appearance and Speech Questionnaire (CHASQ) and CLEFT-Q. The second aim of the study was to investigate patient opinion on the 2 PROMs. DESIGN: Cross-sectional questionnaire study. SETTING: Participants were recruited from a University Hospital. They answered CHASQ and CLEFT-Q either in the hospital or at home. PARTICIPANTS: Thirty-three participants with cleft lip and/or palate, aged 10 to 19 years. MAIN OUTCOME MEASURE: CHASQ and CLEFT-Q. RESULTS: The CHASQ scores and the corresponding CLEFT-Q scores on appearance correlated significantly. Corresponding scores regarding speech did not correlate significantly. A majority, 15 (58%) participants, answered that they liked CLEFT-Q more than CHASQ, 18 participants (69%) thought CHASQ was easier to complete, and 19 (76%) thought CLEFT-Q would better inform health care professionals. CONCLUSION: Both instruments showed strengths and limitations. Clinicians will have to consider each instrument's respective qualities when choosing to implement either PROM.
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Fenda Labial , Fissura Palatina , Estudos Transversais , Audição , Humanos , Fala , Inquéritos e Questionários , SuéciaRESUMO
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.
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Fenda Labial/cirurgia , Fissura Palatina/cirurgia , Sistema de Registros , Adolescente , Criança , Pré-Escolar , Feminino , Humanos , Masculino , Ortodontia , Reprodutibilidade dos Testes , Fala , Suécia , Adulto JovemRESUMO
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.
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Fenda Labial , Fissura Palatina , Insuficiência Velofaríngea , Pré-Escolar , Humanos , Indicadores e Reagentes , Sistema de Registros , Reprodutibilidade dos Testes , Estudos Retrospectivos , Fala , SuéciaRESUMO
OBJECTIVES: The present study validated data that had been reported to the Swedish Quality Registry for Cleft Lip and Palate (CLP) under new requirements from 2016, when use of the 5-year-old (5YO) and the Modified Huddart and Bodenham (MHB) indices for rating occlusion in children born with unilateral CLP (UCLP) was introduced. MATERIALS AND METHODS: The sample included blinded study casts (n = 97) and photos (n = 4) of 5-year-old children who had been born with UCLP in 2009-2011 and were enrolled at one of six cleft centres in Sweden. Fourteen orthodontists from the centres assessed the patients (n = 101) using the 5YO and the MHB indices. Median 5YO and MHB scores of the 14 assessments were compared with original registry data (n = 61). Each centre devised code keys to protect the identities of their patients in the registry. RESULTS: Interrater agreement among the 14 orthodontists was good for the 5YO index (quadratic-weighted kappa: 0.72-0.92) and the MHB index (intraclass correlation coefficient: 0.991-0.994). Comparisons of median 5YOs for each identifiable child with their registry data (n = 61) found total agreement for 70.5 per cent. Comparisons between median MHBs and registry data showed very good or good agreement in 93.4 per cent of the cases. LIMITATIONS: Two teams lost their code keys, which reduced the sample to 61 patients. CONCLUSIONS: The dentoalveolar outcome data in the CLP registry was trustworthy. There was good agreement among the Swedish cleft teams assessing the 5YO and MHB indices in children born with UCLP at age 5 years.
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Fenda Labial , Fissura Palatina , Arco Dental , Sistema de Registros , Pré-Escolar , Fenda Labial/cirurgia , Fissura Palatina/cirurgia , Arco Dental/anatomia & histologia , Arco Dental/cirurgia , Humanos , Reprodutibilidade dos Testes , Suécia , Resultado do TratamentoRESUMO
OBJECTIVE: To test the reliability of the speech data in the Swedish quality registry for cleft lip and palate. DESIGN: Retrospective study. SETTING: Primary care university hospital. PARTICIPANTS: Ninety-four children born with cleft palate with or without cleft lip between 2005 and 2009 who had been assessed and registered in the quality registry at the age of 5 years. MAIN OUTCOME MEASURES: Data in the registry on percent oral consonants correct, percent oral errors, percent nonoral errors, perceived velopharyngeal function (PVPF), and intelligibility were compared with results based on reassessments by 3 independent raters from audio recordings. Agreement was calculated by the intraclass correlation coefficient (ICC), quadratic weighted kappa, and percentage agreement. RESULTS: Absolute agreement calculated by average measures ICC for percent oral consonants correct, percent oral errors, and percent nonoral errors was above >0.90. Single measures ICC for percent oral consonants correct was 0.82, for percent oral errors 0.69, and for percent nonoral errors 0.83. The kappa coefficient for PVPF was 0.5 to 0.59 and for intelligibility 0.65 to 0.77. Exact percentage agreement for PVPF was 33% and for intelligibility 47.8%. CONCLUSIONS: The data on oral consonants correct and nonoral errors in the quality registry seem to be reliable. The data on oral errors, PVPF, and intelligibility should be interpreted with caution. If differences among treatment centers are detected, one should go back and examine the collected raw data before drawing any definitive conclusions about treatment outcome.
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Fenda Labial , Fissura Palatina , Fala , Criança , Humanos , Sistema de Registros , Reprodutibilidade dos Testes , Estudos Retrospectivos , SuéciaRESUMO
OBJECTIVE: To evaluate speech in 5-year-olds with cleft palate with or without cleft lip (CP±L) treated with primary palatal surgery in 1 stage with muscle reconstruction according to Sommerlad at about 12 months of age. DESIGN: Retrospective study. SETTING: Primary care university hospital. PARTICIPANTS: Eight 5-year-olds with cleft soft palate (SP), 22 with cleft soft/hard palate (SHP), 33 with unilateral cleft lip and palate, and 17 with bilateral CLP (BCLP). MAIN OUTCOME MEASURES: Percent oral consonants correct (POCC), percent consonants correct adjusted for age (PCC-A), percent oral errors, percent nonoral errors, and variables related to velopharyngeal function were analyzed from assessments of audio recordings by 3 independent speech-language pathologists. RESULTS: The median POCC was 75.4% (range: 22.7%-98.9%), median PCC-A 96.9% (range: 36.9%-100%), median percent oral errors 3.4% (range: 0%-40.7%), and median percent nonoral errors 0% (range: 0%-20%), with significantly poorer results in children with more extensive clefts. The SP group had significantly less occurrence of audible nasal air leakage than the SHP and the BCLP groups. Before age 5 years, 1.3% of the children underwent fistula surgery and 6.3% secondary speech improving surgery. At age 5 years, 15% of the total group was perceived as having incompetent velopharyngeal function. CONCLUSIONS: Speech was poorer in many children with more extensive clefts. Children with CP±L had poorer speech compared to normative data of peers without CP±L, but the results indicated relatively good speech compared to speech of children with CP±L in previous studies.
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Fenda Labial/fisiopatologia , Fissura Palatina/fisiopatologia , Fissura Palatina/cirurgia , Distúrbios da Fala/fisiopatologia , Pré-Escolar , Fenda Labial/cirurgia , Feminino , Humanos , Masculino , Estudos Retrospectivos , Medida da Produção da FalaRESUMO
BACKGROUND: In recent years, analyses of cleft palate speech based on phonetic transcriptions have become common. However, the results vary considerably among different studies. It cannot be excluded that differences in assessment methodology, including the recording medium, influence the results. AIMS: To compare phonetic transcriptions from audio and audio/video recordings of cleft palate speech by means of outcomes of per cent correct consonants (PCC) and differences in consonant transcriptions. METHODS & PROCEDURES: Thirty-two 3-year-olds born with cleft palate were audio and audio/video recorded while performing a single-word test by picture naming. The recordings were transcribed according to the International Phonetic Alphabet. The transcriptions from the audio and the audio/video recordings were analysed with regard to PCC, differences in the use of phonetic consonant symbols and the use of diacritics. OUTCOMES & RESULTS: PCC was higher when the calculation was based on transcriptions from audio recordings than when based on audio/video recordings. No such differences were seen when age-appropriate consonant processes were scored as correct. Consonants were not excluded as often in the transcriptions from audio/video recordings as they were in the transcriptions from audio recordings, and more target consonants were transcribed as dental/alveolar in the transcriptions from audio/video recordings and palatal/velar/uvular in the transcriptions from audio recordings than vice versa. Further, interdental articulation, linguolabial articulation and audible nasal air leakage were more common in the transcriptions from audio/video recordings than in those from audio recordings. All these differences were statistically significant. CONCLUSIONS & IMPLICATIONS: Phonetic transcription is influenced to some extent by visual cues. However, as long as age-appropriate articulatory and phonological simplification processes are scored as correct when evaluating consonant production in the speech of young children born with cleft palate using phonetic transcription, the recording medium does not seem to affect the results.
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Acústica , Fissura Palatina/diagnóstico , Fonética , Medida da Produção da Fala/métodos , Patologia da Fala e Linguagem/métodos , Fala , Gravação em Vídeo , Qualidade da Voz , Pré-Escolar , Fissura Palatina/fisiopatologia , Fissura Palatina/psicologia , Sinais (Psicologia) , Feminino , Humanos , Masculino , Variações Dependentes do Observador , Valor Preditivo dos Testes , Reprodutibilidade dos Testes , Percepção VisualRESUMO
BACKGROUND: Research regarding expressive language performance in children born with cleft palate is sparse. The relationship between articulation/phonology and expressive language skills also needs to be further explored. AIMS: To investigate verbal competence in narrative retelling in 5-year-old children born with unilateral cleft lip and palate (UCLP) and its possible relationship with articulation/phonology at 3 and 5 years of age. METHODS & PROCEDURES: A total of 49 children, 29 with UCLP treated according to three different procedures for primary palatal surgery and a comparison group of 20 children (COMP), were included. Longitudinally recorded audio files were used for analysis. At ages 3 and 5, the children were presented with a single-word test of word naming and at age 5 also the Bus Story Test (BST). The BST was assessed according to a test manual. The single-word test was phonetically transcribed and the percentage of consonants correct adjusted for age (PCC-A) was calculated. Differences regarding the BST results within the UCLP group were analysed. The results were compared with the results of the COMP group, and also with norm values. In addition, the relationship between the results of the BST and the PCC-A scores at ages 3 and 5 years was analysed. OUTCOMES & RESULTS: No significant group differences or correlations were found. However, 65.5% of the children in the UCLP group had an information score below 1 standard deviation from the norm value compared with 30% in the COMP group. CONCLUSIONS: A larger proportion of children in the UCLP group than in the COMP group displayed problems with retelling but the differences between the two groups were not significant. There was no association between the BST results in the children with UCLP and previous or present articulatory/phonological competence. Since group size was small in both groups, the findings need to be verified in a larger study.
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Transtornos da Articulação/diagnóstico , Fenda Labial/diagnóstico , Fissura Palatina/diagnóstico , Narração , Testes de Articulação da Fala , Medida da Produção da Fala , Comportamento Verbal , Pré-Escolar , Feminino , Humanos , Masculino , Fonação , Fonética , Estudos Prospectivos , Estudos Retrospectivos , Acústica da Fala , Inteligibilidade da Fala , SuéciaRESUMO
BACKGROUND: Approximately 50% of children born with cleft palate present speech difficulties around 3 years of age, and several studies report on persisting phonological problems after palatal closure. However, studies on early phonology related to cleft palate are few and have so far mainly been carried out on English-speaking children. Studies on phonology related to cleft palate in languages other than English are also warranted. AIMS: To assess phonology in Swedish-speaking children born with and without unilateral cleft lip and palate (UCLP) at 3 years of age, and to identify variables at 18 months that are associated with restricted phonology at age 3 years. METHODS & PROCEDURES: Eighteen consecutive children born with UCLP and 20 children without cleft lip and palate were included. Transcriptions of audio recordings at 18 months and 3 years were used. Per cent correct consonants adjusted for age (PCC-A), the number of established phonemes, and phonological simplification processes at 3 years were assessed and compared with different aspects of consonant inventory at 18 months. OUTCOMES & RESULTS: PCC-A, the number of established phonemes, and the total number of phonological processes differed significantly at 3 years between the two groups. Total number of oral consonants, oral stops, dental/alveolar oral stops and number of different oral stops at 18 months correlated significantly with PCC-A at 3 years in the UCLP group. CONCLUSIONS & IMPLICATIONS: As a group, children born with UCLP displayed deviant phonology at 3 years compared with peers without cleft lip and palate. Measures of oral consonant and stop production at 18 months might be possible predictors for phonology at 3 years in children born with cleft palate.
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Transtornos da Articulação/diagnóstico , Fenda Labial/reabilitação , Fissura Palatina/reabilitação , Fonética , Distúrbios da Fala/diagnóstico , Fala , Transtornos da Articulação/etiologia , Pré-Escolar , Fenda Labial/complicações , Fissura Palatina/complicações , Feminino , Audição , Transtornos da Audição/complicações , Transtornos da Audição/diagnóstico , Humanos , Lactente , Masculino , Distúrbios da Fala/etiologia , Medida da Produção da Fala , Fonoterapia , SuéciaRESUMO
Objective : To describe and compare speech and phonology at age 3 years in children born with unilateral complete cleft lip and palate treated with three different methods for primary palatal surgery. Design : Prospective study. Setting : Primary care university hospitals. Participants : Twenty-eight Swedish-speaking children born with nonsyndromic unilateral complete cleft lip and palate. Interventions : Three methods for primary palatal surgery: two-stage closure with soft palate closure between 3.4 and 6.4 months and hard palate closure at mean age 12.3 months (n = 9) or 36.2 months (n = 9) or one-stage closure at mean age 13.6 months (n = 10). Main Outcome Measures : Based on independent judgments performed by two speech-language pathologists from standardized video recordings: percent correct consonants adjusted for age, percent active cleft speech characteristics, total number of phonological processes, number of different phonological processes, hypernasality, and audible nasal air leakage. The hard palate was unrepaired in nine of the children treated with two-stage closure. Results : The group treated with one-stage closure showed significantly better results than the group with an unoperated hard palate regarding percent active cleft speech characteristics and total number of phonological processes. Conclusions : Early primary palatal surgery in one or two stages did not result in any significant differences in speech production at age 3 years. However, children with an unoperated hard palate had significantly poorer speech and phonology than peers who had been treated with one-stage palatal closure at about 13 months of age.
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Fenda Labial/cirurgia , Fissura Palatina/cirurgia , Fonação , Distúrbios da Fala/epidemiologia , Fala , Pré-Escolar , Feminino , Hospitais Universitários , Humanos , Masculino , Procedimentos Cirúrgicos Otorrinolaringológicos , Estudos Prospectivos , Medida da Produção da Fala , Suécia/epidemiologiaRESUMO
Recently, Ombashi et al. published a systematic review aiming to identify the pitfalls in the development and implementation as well as factors influencing long-term success of a multidisciplinary, international registry for cleft care on a global scale. The purpose of this letter to the editor is to highlight that the review failed to include the Swedish quality registry for patients born with cleft lip and palate, which fulfils the inclusion criteria. The Swedish cleft lip and palate registry is multidisciplinary, has a high coverage and reporting degree, and most outcome measures have been checked for reliability and validity. It is regularly used for open comparisons between treatment centers. Several research studies have been published based on the Swedish cleft lip and palate registry, and more are ongoing. The information we provide about the Swedish cleft lip and palate registry complements and expands the information of the results reported by Ombashi et al. in their research.
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Fenda Labial , Fissura Palatina , Doenças Raras , Sistema de Registros , Humanos , SuéciaRESUMO
INTRODUCTION: Speech in children with cleft palate can be affected by velopharyngeal dysfunction, which persists after primary palate repair. The incidence of surgery to correct velopharyngeal dysfunction in this patient group has previously been reported as 2.6-37%. We aimed to investigate the incidence of velopharyngeal dysfunction surgery in Swedish children with cleft palate and to examine potential associations of independent variables with this incidence. METHODS: In this cohort study, we analysed data from the Swedish cleft lip and palate quality registry for 1093 children with cleft palate with or without cleft lip. Kaplan-Meier analysis was used to estimate the risk of having velopharyngeal dysfunction surgery. Multivariable Cox proportional hazards models were used to estimate the associated effect of cleft subtype, additional diagnoses, gender, and age at and number of stages for primary palate repair on the primary outcome. RESULTS: The risk of having velopharyngeal dysfunction surgery was 25.6%. Complete primary palate repair after the age of 18 months or in more than one stage was associated with a higher risk, but it could not be determined which of these was the more significant factor. Cleft soft palate was associated with a significantly lower risk than other cleft subtypes. CONCLUSIONS: Primary palate repair at a higher age or in more than one stage may increase the risk of having velopharyngeal dysfunction surgery. Further analysis of potential unknown confounding factors and the association between the incidence of velopharyngeal dysfunction and surgery to correct this condition is needed.
Assuntos
Fenda Labial , Fissura Palatina , Insuficiência Velofaríngea , Criança , Humanos , Lactente , Fissura Palatina/complicações , Fissura Palatina/cirurgia , Fenda Labial/complicações , Fenda Labial/epidemiologia , Fenda Labial/cirurgia , Estudos de Coortes , Suécia/epidemiologia , Incidência , Insuficiência Velofaríngea/epidemiologia , Insuficiência Velofaríngea/etiologia , Insuficiência Velofaríngea/cirurgia , Resultado do Tratamento , Palato Mole , FalaRESUMO
Cleft lip and palate (CL/P) is the most common congenital craniofacial malformation and is often associated with additional diagnoses. The purpose of this study was to explore the cumulative five-year incidence of additional diagnoses for patients with cleft lip and palate. Further aims were, type of cleft and type of additional diagnose and to validate CLP registry data on additional diagnoses. Data from the CLP registry regarding children with CL/P in the Southern Health Care Region were retrieved and based on the registry, participants were selected. A review of medical records of participants born 2006-2016 was performed and data regarding participant characteristics and additional diagnoses were collected. Of the 250 participants included in the review of medical records, 90 participants (36%) had an additional diagnosis. Of the total number of identified additional diagnoses (n = 137), cardiovascular system (20.4%) and extremities and skeletal system (17.5%) were the most prevalent categories. The comparison between medical records and the CLP registry of all children showed a 14.4 percentage points higher incidence of additional diagnoses in the medical records. Roughly every third child received an additional diagnosis and diagnoses related to the cardiovascular system were the most frequent. This study also shows that additional diagnoses were under-reported in the CLP registry. Future research is necessary to strengthen associations of additional diagnoses to CL/P.