| 1. |
- Semb, Gunvor, et al.
(författare)
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A Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 1. Planning and management.
- 2017
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Ingår i: Journal of Plastic Surgery and Hand Surgery. - : Taylor & Francis. - 2000-656X .- 2000-6764. ; 51:1, s. 2-13
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Tidskriftsartikel (refereegranskat)abstract
- BACKGROUND AND AIMS: Longstanding uncertainty surrounds the selection of surgical protocols for the closure of unilateral cleft lip and palate, and randomised trials have only rarely been performed. This paper is an introduction to three randomised trials of primary surgery for children born with complete unilateral cleft lip and palate (UCLP). It presents the protocol developed for the trials in CONSORT format, and describes the management structure that was developed to achieve the long-term engagement and commitment required to complete the project.METHOD: Ten established national or regional cleft centres participated. Lip and soft palate closure at 3-4 months, and hard palate closure at 12 months served as a common method in each trial. Trial 1 compared this with hard palate closure at 36 months. Trial 2 compared it with lip closure at 3-4 months and hard and soft palate closure at 12 months. Trial 3 compared it with lip and hard palate closure at 3-4 months and soft palate closure at 12 months. The primary outcomes were speech and dentofacial development, with a series of perioperative and longer-term secondary outcomes.RESULTS: Recruitment of 448 infants took place over a 9-year period, with 99.8% subsequent retention at 5 years.CONCLUSION: The series of reports that follow this introductory paper include comparisons at age 5 of surgical outcomes, speech outcomes, measures of dentofacial development and appearance, and parental satisfaction. The outcomes recorded and the numbers analysed for each outcome and time point are described in the series.TRIAL REGISTRATION: ISRCTN29932826.
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| 2. |
- 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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| 3. |
- 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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| 4. |
- Lohmander, Anette, et al.
(författare)
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Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 4. Speech outcomes in 5-year-olds - velopharyngeal competency and hypernasality
- 2017
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Ingår i: Journal of Plastic Surgery and Hand Surgery. - : TAYLOR & FRANCIS LTD. - 2000-656X .- 2000-6764. ; 51:1, s. 27-37
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Tidskriftsartikel (refereegranskat)abstract
- Background and aim: Adequate velopharyngeal function and speech are main goals in the treatment of cleft palate. The objective was to investigate if there were differences in velopharyngeal competency (VPC) and hypernasality at age 5 years in children with unilateral cleft lip and palate (UCLP) operated on with different surgical methods for primary palatal repair. A secondary aim was to estimate burden of care in terms of received additional secondary surgeries and speech therapy. Design: Three parallel group, randomised clinical trials were undertaken as an international multicentre study by 10 cleft teams in five countries: Denmark, Finland, Sweden, Norway, and the UK. Methods: Three different surgical protocols for primary palatal repair were tested against a common procedure in the total cohort of 448 children born with a non-syndromic UCLP. Speech audio and video recordings of 391 children (136 girls, 255 boys) were available and perceptually analysed. The main outcome measures were VPC and hypernasality from blinded assessments. Results: There were no statistically significant differences between the prevalences in the arms in any of the trials. VPC: Trial 1, A: 58%, B: 61%; Trial 2, A: 57%, C: 54%; Trial 3, A: 35%, D: 51%. No hypernasality: Trial 1, A: 54%, B: 44%; Trial 2, A: 47%, C: 51%; Trial 3, A: 34%, D: 49%. Conclusions: No differences were found regarding VPC and hypernasality at age 5 years after different methods for primary palatal repair. The burden of care in terms of secondary pharyngeal surgeries, number of fistulae, and speech therapy visits differed.
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| 5. |
- Lohmander, Anette, et al.
(författare)
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Validity of auditory perceptual assessment of velopharyngeal function and dysfunction - the VPC-Sum and the VPC-Rate
- 2017
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Ingår i: Clinical Linguistics & Phonetics. - : TAYLOR & FRANCIS INC. - 0269-9206 .- 1464-5076. ; 31:7-9, s. 589-597
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Tidskriftsartikel (refereegranskat)abstract
- 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 amp;lt; 0.001), and a moderately significant positive correlation between VPC-Sum and VPC-Rate (Rho 0.698, P amp;lt; 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.
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| 6. |
- Persson, Christina, 1959, et al.
(författare)
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Scandcleft Project Trial 3: Comparison of Speech Outcomes in Relation to Sequence in 2-Stage Palatal Repair Procedures in 5-Year-Olds With Unilateral Cleft Lip and Palate.
- 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. ; 57:3, s. 352-363
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Tidskriftsartikel (refereegranskat)abstract
- To compare speech outcome following different sequencing of hard and soft palate closure between arms and centers within trial 3 and compare results to peers without cleft palate.A prospective randomized clinical trial.Two Norwegian and 2 British centers.One hundred thirty-six 5-year-olds with unilateral cleft lip and palate were randomized to either lip and soft palate closure at 3 to 4 months and hard palate closure at 12 months (arm A) or lip and hard palate closure at 3 to 4 months and soft palate closure at 12 months (arm D).A composite measure of velopharyngeal competence (VPC), overall assessment of VPC from connected speech (VPC-Rate). Percentage of consonants correct (PCC), active cleft speech characteristics (CSCs), subdivided by oral retracted and nonoral errors, and developmental speech characteristics (DSCs).Across the trial, 47% had VPC, with no statistically significant difference between arms within or across centers. Thirty-eight percent achieved a PCC score of >90%, with no difference between arms or centers. In one center, significantly more children in arm A produced ≥3 active CSCs (P < .05). Across centers, there was a statistically significant difference in active CSCs (arm D), oral retracted CSCs (arm D), and DSCs (arms A and D).Less than half of the 5-year-olds achieved VPC and around one-third achieved age-appropriate PCC scores. Cleft speech characteristics were more common in arm A, but outcomes varied within and across centers. Thus, outcome of the same surgical method can vary substantially across centers.
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| 7. |
- Sondergaard, Elisabeth, et al.
(författare)
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Problems and challenges in relation to the treatment of patients with multimorbidity: General practitioners views and attitudes
- 2015
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Ingår i: Scandinavian Journal of Primary Health Care. - : TAYLOR and FRANCIS LTD. - 0281-3432 .- 1502-7724. ; 33:2, s. 121-126
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Tidskriftsartikel (refereegranskat)abstract
- Objective. To explore views and attitudes among general practitioners (GPs) and researchers in the field of general practice towards problems and challenges related to treatment of patients with multimorbidity. Setting. A workshop entitled Patients with multimorbidity in general practice held during the Nordic Congress of General Practice in Tampere, Finland, 2013. Subjects. A total of 180 GPs and researchers. Design. Data for this summary report originate from audio-recorded, transcribed verbatim plenary discussions as well as 76 short questionnaires answered by attendees during the workshop. The data were analysed using framework analysis. Results. (i) Complex care pathways and clinical guidelines developed for single diseases were identified as very challenging when handling patients with multimorbidity; (ii) insufficient cooperation between the professionals involved in the care of multimorbid patients underlined the GPs impression of a fragmented health care system; (iii) GPs found it challenging to establish a good dialogue and prioritize problems with patients within the timeframe of a normal consultation; (iv) the future role of the GP was discussed in relation to diminishing health inequality, and current payment systems were criticized for not matching the treatment patterns of patients with multimorbidity. Conclusion. The participants supported the development of a future research strategy to improve the treatment of patients with multimorbidity. Four main areas were identified, which need to be investigated further to improve care for this steadily growing patient group.
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| 8. |
- Willadsen, Elisabeth, et al.
(författare)
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A software program to assist coding of prelinguistic vocalizations in real time.
- 2018
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Ingår i: Clinical linguistics & phonetics. - : Informa UK Limited. - 1464-5076 .- 0269-9206. ; 32:10, s. 972-978
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Tidskriftsartikel (refereegranskat)abstract
- Numerous studies have confirmed that prelinguistic utterances are precursors to speech, and there is ample evidence that, for example, frequency of canonical syllables and syllable inventory size correlate with speech and language measures at older ages. Traditionally, prelinguistic utterances have been assessed by phonetic transcription which is difficult and time-consuming in infants. Recently, a more time-efficient methodology to assess prelinguistic utterances in real time, called naturalistic listening, was developed (Ramsdell et al., 2012). In a large international NIDCR-funded randomized controlled trial, Timing of Primary Surgery for with Cleft Palate (TOPS), including many coders, a software program (TimeStamper) was developed to assist in coding of prelinguistic vocalizations in real time, to ensure consistency of the coding procedures. Coders upload a video (or audio) file and watch and listen to the recording in real time without any possibility of pausing or taking notes. In real time, the coder registers each speech-like syllable as canonical or non-canonical. TimeStamper automatically calculates the percentage of canonical syllables of all syllables registered (canonical babbling ratio). At the end of a recording, TimeStamper assists in assessing presence/absence of canonical babbling and syllable inventory size. The software is presented and instructions for free access are provided.
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| 9. |
- Willadsen, Elisabeth, et al.
(författare)
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Assessment of prelinguistic vocalizations in real time: a comparison with phonetic transcription and assessment of inter-coder-reliability
- 2020
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Ingår i: Clinical Linguistics and Phonetics. - : Informa UK Limited. - 0269-9206 .- 1464-5076. ; 34:7, s. 593-616
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Tidskriftsartikel (refereegranskat)abstract
- © 2019, © 2019 Taylor & Francis Group, LLC. This study investigated reliability of naturalistic listening in real time (NLRT) compared to phonetic transcription. Speech pathology students with brief training in NLRT assessed prelinguistic syllable inventory size and specific syllable types in typically developing infants. A second study also examined inter-coder reliability for canonical babbling, canonical babbling ratio and presence of oral stops in syllable inventory of infants with cleft palate, by means of NLRT. In study 1, ten students independently assessed prelinguistic samples of five 12-month-old typically developing infants using NLRT and phonetic transcription. Coders assessed syllable inventory size as more than twice as large using phonetic transcription as NLRT. Results showed a strong correlation between NLRT and phonetic transcription (syllables with more than five occurrences) for syllable inventory size (r=.60; p <.001). The methods showed similar results for inter-coder reliability of specific syllable types. In study 2, three other students assessed prelinguistic samples of twenty-eight 12-month-old infants with cleft palate by means of NLRT. Results revealed perfect inter-coder agreement for presence/absence of canonical babbling, strong correlations between the three coders’ assessment of syllable inventory size (average r =.83; p <.001), but more inter-coder variability for agreement of specific syllable types. In conclusion, NLRT is a reliable method for assessing prelinguistic measures in infants with and without cleft palate with inter-coder agreement levels comparable to phonetic transcription for specific syllable types.
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| 10. |
- Willadsen, Elisabeth, et al.
(författare)
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Scandcleft Project, Trial 1: Comparison of Speech Outcome in Relation to Timing of Hard Palate Closure in 5-Year-Olds With UCLP.
- 2019
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Ingår i: The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association. - : SAGE Publications. - 1545-1569. ; 56:10, s. 1276-1286
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Tidskriftsartikel (refereegranskat)abstract
- To compare in-depth speech results in Scandcleft Trial 1 as well as reference data from peers without cleft palate (CP).A prospective randomized clinical trial.A Danish and a Swedish CP center.143 of 148 randomized 5-year-olds with unilateral cleft lip and palate. All received lip and velum closure at 4 months, and hard palate closure at 12 months (arm A) or 36 months (arm B).A composite measure based on velopharyngeal competence (VPC) or velopharyngeal incompetence (VPI), an overall assessment of VPC from connected speech (VPC-Rate), Percentage of Consonants Correct (PCC-score), and consonant errors. Speech therapy visits, average hearing thresholds, and secondary pharyngeal surgeries documented burden of treatment.Across the trial, 61.5% demonstrated VPC and 38.5% VPI. Twenty-two percent of participants achieved age appropriate PCC-scores. There were no statistically significant differences between arms or centers for these measures. In the Danish center, arm B: achieved lower PCC-scores (P = .01); obtained PCC-scores without s-errors below 79% (P = .002); produced ≥3 active oral cleft speech characteristics (P = .004) than arm A. In both centers, arm B attended more speech visits.At age 5, differences between centers and treatment arms were not statistically significant for VPC/VPI, but consonant proficiency differed between treatment arms in the Danish center. Poor speech outcomes were seen for both treatment arms. Variations between centers were observed. As the Swedish center had few participants, intercenter comparisons should be interpreted with caution.
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