| 1. |
- Schölin, Johnna, 1970, et al.
(författare)
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Surgical, speech, and hearing outcomes at five years of age in internationally adopted children and Swedish-born children with cleft lip and/or palate
- 2020
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Ingår i: Journal of Plastic Surgery and Hand Surgery. - : Medical Journals Sweden AB. - 2000-656X .- 2000-6764. ; 54:1, s. 6-13
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Tidskriftsartikel (refereegranskat)abstract
- Internationally adopted children (IAC) with a cleft lip and/or palate (CL/P) tend to arrive with un-operated palates at an age at which their Swedish-born peers have completed their primary palate surgery. Our aim of the present study was to analyze surgical, speech and hearing outcomes of IAC at age 5 and compare with those of a matched group of Swedish-born children. Fifty children with CL/P born in 1994-2005 participated in the study. Twenty-five IAC were matched according to age, sex and cleft type with 25 Swedish-born children. Audio recordings were perceptually analyzed by two experienced, blinded speech-language pathologists. Hearing and speech statuses were evaluated on the same day for all children. Surgical timing and complications as in fistulas and requirement for secondary velopharyngeal (VP) surgery, speech evaluation results, and present hearing status were analyzed for all children of age 5 years. Results showed that primary palatal surgery was delayed by a mean of 21 months in IAC. IAC had a higher prevalence of velopharyngeal impairment that was statistically significant, a higher fistula rate, and experienced more secondary surgery than Swedish-born peers. Hearing loss due to middle ear disease was slightly more common among IAC, whereas the rate of treatment with tympanostomy tubes was similar between the two groups. In conclusion, IAC with CL/P represent a challenge for CL/P teams because of the heterogeneous nature of the patient group and difficulties associated with delayed treatment, and the results show the importance of close follow-up over time.
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| 2. |
- Gamble, Carrol, et al.
(författare)
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Timing of Primary Surgery for Cleft Palate.
- 2023
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Ingår i: The New England journal of medicine. - : Massachusetts Medical Society. - 1533-4406 .- 0028-4793. ; 389:9, s. 795-807
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Tidskriftsartikel (refereegranskat)abstract
- 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.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.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.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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| 3. |
- Larsson, AnnaKarin, 1975, et al.
(författare)
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Internationally Adopted Children With Unilateral Cleft Lip and Palate-Consonant Proficiency and Perceived Velopharyngeal Competence at the Age of 5.
- 2020
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Ingår i: The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association. - : SAGE Publications. - 1545-1569. ; 57:7, s. 849-859
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Tidskriftsartikel (refereegranskat)abstract
- To compare consonant proficiency, consonant errors, and the perceived velopharyngeal (VP) competence in internationally adopted (IA) children with unilateral cleft lip and palate (UCLP) and nonadopted (NA) children with the same cleft-palate type at age 5.Case-control study based on phonetic transcriptions of standardized speech recordings of 5-year-olds at a tertiary hospital.Twenty-five IA children were compared to 20 NA children. All consecutive patients at a cleft lip and palate center participated.Consonant proficiency was measured using percentage consonants correct, percentage consonants correct-adjusted for age, percentage correct place, percentage correct manner, and consonant inventory. Cleft speech characteristics (CSCs), developmental speech characteristics (DSCs), and the perceived VP competence were also measured.The IA children had significantly lower values for all consonant proficiency variables (p < .05) and a smaller consonant inventory (p = .001) compared to the NA children. The IA children had a higher frequency of CSCs (IA = 84%, NA = 50%, p < .05) and DSCs (IA = 92%, NA = 65%, p = .057), and twice as many IA children as NA children had perceived VP incompetence (IA = 52%, NA = 25%, p = .17).Severe speech disorder was more common in IA children than in NA children at age 5. Most importantly, the speech disorders seem to be not only cleft-related. More detailed speech assessments with a broader focus are needed for IA children with UCLP. Longitudinal studies are recommended to further investigate the impact of speech difficulties in IA children's daily lives.
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| 4. |
- Larsson, AnnaKarin, 1975, et al.
(författare)
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Persisting speech difficulties at 7-8 years of age - a longitudinal study of speech production in internationally adopted children with cleft lip and palate
- 2024
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Ingår i: Logopedics Phoniatrics Vocology. - : Informa UK Limited. - 1401-5439 .- 1651-2022. ; 49:1, s. 1-10
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Tidskriftsartikel (refereegranskat)abstract
- Aim To longitudinally investigate speech production (consonant proficiency, consonant errors and perceived velopharyngeal competence) in 17 internationally adopted (IA) children with unilateral cleft lip and palate (UCLP) at three time points. Method Consonant proficiency (percent consonants correct, consonant inventory) and number and type of consonant errors were calculated based on blind phonetic transcriptions of words from the Swedish Test of Articulation and Nasality (SVANTE). Velopharyngeal competence was perceptually rated by three blinded experienced speech-language pathologists at the ages of 3, 5 and 7-8 years. Results A significant positive development of speech production was found, although most children still scored very low for consonant proficiency at the age of 7-8 compared with normative values: the median for percent consonants correct was 79.7 and many children still had persisting cleft-related and developmental consonant errors. At the age of 7-8, almost half of the children were rated as having a competent velopharyngeal function and only three as having an incompetent velopharyngeal function. Conclusion Persisting speech difficulties at school age in IA children with UCLP were found in the present study, which is one of the very few longitudinal studies. Our results highlight the need for detailed follow-up of speech production in clinical settings. Speech disorders may have a severe impact on a child's intelligibility and participation with peers, and there is a need for more studies investigating the actual everyday effect of the difficulties found.
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| 5. |
- Persson, Christina, 1959, et al.
(författare)
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Longitudinal Speech Outcome at 5 and 10 Years in UCLP: Influence of Speech Therapy and Secondary Velopharyngeal Surgery
- 2024
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Ingår i: CLEFT PALATE CRANIOFACIAL JOURNAL. - : SAGE PUBLICATIONS INC. - 1055-6656 .- 1545-1569.
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Tidskriftsartikel (refereegranskat)abstract
- Objective: To investigate speech development of children aged 5 and 10 years with repaired unilateral cleft lip and palate (UCLP) and identify speech characteristics when speech proficiency is not at 'peer level' at 10 years. Estimate how the number of speech therapy visits are related to speech proficiency at 10 years, and what factors are predictive of whether a child's speech proficiency at 10 years is at 'peer level' or not. Design: Longitudinal complete datasets from the Scandcleft project Participants: 320 children from nine cleft palate teams in five countries, operated on with one out of four surgical methods. Interventions: Secondary velopharyngeal surgery (VP-surgery) and number of speech therapy visits (ST-visits), a proxy for speech intervention. Main Outcome Measures: 'Peer level' of percentage of consonants correct (PCC, > 91%) and the composite score of velopharyngeal competence (VPC-Sum, 0-1). Results: Speech proficiency improved, with only 23% of the participants at 'peer level' at 5 years, compared to 56% at 10 years. A poorer PCC score was the most sensitive marker for the 44% below 'peer level' at 10-year-of-age. The best predictor of 'peer level' speech proficiency at 10 years was speech proficiency at 5 years. A high number of ST-visits received did not improve the probability of achieving 'peer level' speech, and many children seemed to have received excessive amounts of ST-visits without substantial improvement. Conclusions: It is important to strive for speech at 'peer level' before age 5. Criteria for speech therapy intervention and for methods used needs to be evidence-based.
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| 6. |
- Willadsen, E., et al.
(författare)
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Scandcleft randomized trials of primary surgery for unilateral cleft lip and palate: Speech proficiency at 10 years of age
- 2023
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Ingår i: International journal of language and communication disorders. - : WILEY. - 1368-2822 .- 1460-6984. ; 58:3, s. 892-909
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Tidskriftsartikel (refereegranskat)abstract
- Background & AimTo assess consonant proficiency and velopharyngeal function in 10-year-old children born with unilateral cleft lip and palate (UCLP) within the Scandcleft project. Methods & ProceduresThree parallel group, randomized, clinical trials were undertaken as an international multicentre study by nine cleft teams in five countries. Three different surgical protocols for primary palate repair (Arm B-Lip and soft palate closure at 3-4 months, hard palate closure at 36 months, Arm C-Lip closure at 3-4 months, hard and soft palate closure at 12 months, and Arm D-Lip closure at 3-4 months combined with a single-layer closure of the hard palate using a vomer flap, soft palate closure at 12 months) were tested against a common procedure (Arm A-Lip and soft palate closure at 3-4 months followed by hard palate closure at 12 months) in the total cohort of 431 children born with a non-syndromic UCLP. Speech audio and video recordings of 399 children were available and perceptually analysed. Percentage of consonants correct (PCC) from a naming test, an overall rating of velopharyngeal competence (VPC) (VPC-Rate), and a composite measure (VPC-Sum) were reported. Outcomes & ResultsThe mean levels of consonant proficiency (PCC score) in the trial arms were 86-92% and between 58% and 83% of the children had VPC (VPC-Sum). Only 50-73% of the participants had a consonant proficiency level with their peers. Girls performed better throughout. Long delay of the hard palate repair (Arm B) indicated lower PCC and simultaneous hard and soft palate closure higher (Arm C). However, the proportion of participants with primary VPC (not including velopharyngeal surgeries) was highest in Arm B (68%) and lowest in Arm C (47%). Conclusions & ImplicationsThe speech outcome in terms of PCC and VPC was low across the trials. The different protocols had their pros and cons and there is no obvious evidence to recommend any of the protocols as superior. Aspects other than primary surgical method, such as time after velopharyngeal surgery, surgical experience, hearing level, language difficulties and speech therapy, need to be thoroughly reviewed for a better understanding of what has affected speech outcome at 10 years. WHAT THIS PAPER ADDSWhat is already known on the subjectSpeech outcomes at 10 years of age in children treated for UCLP are sparse and contradictory. Previous studies have examined speech outcomes and the relationship with surgical intervention in 5-year-olds. What this study adds to the existing knowledgeSpeech outcomes based on standardized assessment in a large group of 10-year-old children born with UCLP and surgically treated according to different protocols are presented. While speech therapy had been provided, a large proportion of the children across treatment protocols still needed further speech therapy. What are the potential or actual clinical implications of this work?Aspects other than surgery and speech function might add to the understanding of what affects speech outcome. Effective speech therapy should be available for children in addition to primary surgical repair of the cleft and secondary surgeries if needed.
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| 7. |
- Conroy, Elizabeth J, et al.
(författare)
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A randomised controlled trial comparing palate surgery at 6months versus 12months of age (the TOPS trial): a statistical analysis plan.
- 2021
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Ingår i: Trials. - : Springer Science and Business Media LLC. - 1745-6215. ; 22:1
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Tidskriftsartikel (refereegranskat)abstract
- Cleft palate is among the most common birth abnormalities. The success of primary surgery in the early months of life is crucial for successful feeding, hearing, dental development, and facial growth. Over recent decades, age at palatal surgery in infancy has reduced. The Timing Of Primary Surgery for cleft palate (TOPS) trial aims to determine whether, in infants with cleft palate, it is better to perform primary surgery at age 6 or 12months (corrected for gestational age).The TOPS trial is an international, two-arm, parallel group, randomised controlled trial. The primary outcome is insufficient velopharyngeal function at 5years of age. Secondary outcomes, measured at 12months, 3years, and 5years of age, include measures ofspeech development, safety of the procedure, hearing level, middle ear function, dentofacial development, and growth. The analysis approaches for primary and secondary outcomes are described here, as are the descriptive statistics which will be reported. The TOPS protocol has been published previously.This paper provides details of the planned statistical analyses for the TOPS trial and will reduce the risk of outcome reporting bias and data-driven results.ClinicalTrials.gov NCT00993551 . Registered on 9 October 2009.
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| 8. |
- Justin, Parry Weinfeld, et al.
(författare)
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Prevalence of Reading Difficulties in 9-to 10-Year Old Children in Sweden Born With Cleft Palate
- 2022
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Ingår i: Cleft Palate-Craniofacial Journal. - : SAGE Publications. - 1055-6656 .- 1545-1569. ; 59:4, s. 427-435
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Tidskriftsartikel (refereegranskat)abstract
- Objective: To investigate the prevalence of reading difficulties in children born with cleft palate at ages 9 and 10 in Sweden. Design: Using a cross-sectional design, a parental questionnaire assessing dyslexia-like reading difficulties (Short Dyslexia Scale, SDS) was administered together with separate questions regarding background data. Participants: Families with a child born with overt cleft palate with or without cleft lip in 4 regions of Sweden. A total of 245 families were approached of which 138 families responded. Data from 136 (56%) were complete with information on cleft type and could be analyzed. Results: Twenty-two percent (95% CI, 15-30) of the whole study group displayed risk for dyslexic reading difficulties on the SDS corresponding to the 7th to 10th percentiles in the population. Children with cleft palate only had a significantly higher prevalence of reading difficulties (37%) compared to children with unilateral cleft palate (19%) and bilateral cleft palate (10%). The frequency of reading difficulties in participants with comorbidity was 32%. Among a subgroup with reported comorbidity in areas of attention, language, and learning problems, there was a 2.5 times higher risk of reading disability compared to participants without this reported comorbidity. Conclusion: The prevalence of reading difficulties in the cleft palate population was higher than in the general population. Results showed that co-occurring difficulties were common in the cleft group and that reading difficulties often appear together with other, co-occurring neurodevelopmental difficulties.
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| 9. |
- Larsson, Anna Karin, et al.
(författare)
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Internationally adopted children with and without a cleft lip and palate showed no differences in language ability at school-age
- 2021
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Ingår i: Acta Pædiatrica. - : Wiley. - 1651-2227 .- 0803-5253. ; 110:1, s. 273-279
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Tidskriftsartikel (refereegranskat)abstract
- 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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| 10. |
- Lundeborg Hammarström, Inger, 1956-, et al.
(författare)
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Scandcleft Project Trial 2—Comparison of Speech Outcome in 1- and 2-Stage Palatal Closure in 5-Year-Olds With UCLP
- 2020
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Ingår i: Cleft Palate-Craniofacial Journal. - : SAGE Publications. - 1055-6656 .- 1545-1569. ; 57:4, s. 458-469
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Tidskriftsartikel (refereegranskat)abstract
- Objective: To investigate in-depth speech results in the Scandcleft Trial 2 with comparisons between surgical protocols and centers and with benchmarks from peers without cleft palate. Design: A prospective randomized clinical trial. Setting: Two Swedish and one Finnish Cleft Palate center. Participants: One hundred twelve participants were 5-years-old born with unilateral cleft lip and palate randomized to either lip repair and soft palate closure at 4 months and hard palate closure at 12 months or lip repair at 3 to 4 months (Arm A), or a closure of both the soft and hard palate at 12 months (Arm C). Main Outcome Measures: A composite measure dichotomized into velopharyngeal competency (VPC) or velopharyngeal incompetency (VPI), overall assessment of velopharyngeal function (VPC-Rate), percentage of consonants correct (PCC score), and consonant errors. In addition, number of speech therapy visits, average hearing thresholds, and secondary surgeries were documented to assess burden of treatment. Results: Across the trial, 53.5% demonstrated VPC and 46.5% VPI with no significant differences between arms or centers. In total, 27% reached age-appropriate PCC scores with no statistically significant difference between the arms. The Finnish center had significantly higher PCC scores, the Swedish centers had higher percentages of oral consonant errors. Number of speech therapy visits was significantly higher in the Finnish center. Conclusion: At age 5, poor speech outcomes with some differences between participating centers were seen but could not be attributed to surgical protocol. As one center had very few participants, the results from that center should be interpreted with caution. © 2019, American Cleft Palate-Craniofacial Association.
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