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.

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.TRIAL REGISTRATION:ISRCTN29932826.

Overall weighted or composite variables for perceptual auditory estimation of velopharyngeal closure or competence have been used in several studies for evaluation of velopharyngeal function during speech. The aim of the present study was to investigate the validity of a composite score (VPC-Sum) and of auditory perceptual ratings of velopharyngeal competence (VPC-Rate). Available VPC-Sum scores and judgments of associated variables (hypernasality, audible nasal air leakage, weak pressure consonants, and non-oral articulation) from 391 5-year olds with repaired cleft palate (the Scandcleft project) were used to investigate content validity, and 339 of these were compared with an overall judgment of velopharyngeal competence (VPC-Rate) on the same patients by the same listeners. Significant positive correlations were found between the VPC-Sum and each of the associated variables (Cronbachs alpha 0.55-0.87, P < 0.001), and a moderately significant positive correlation between VPC-Sum and VPC-Rate (Rho 0.698, P < 0.01). The latter classified cases well when VPC-Sum was dichotomized with 67% predicted velopharyngeal competence and 90% velopharyngeal incompetence. The validity of the VPC-Sum was good and the VPC-Rate a good predictor, suggesting possible use of both measures depending on the objective.

Background and aim: Few studies have explored children's emotional and behavioural reactions to cleft surgery and treatment-related stress. The objective was to investigate parents' evaluations of appearance and treatment outcomes in their 5-year-old child with unilateral cleft lip and palate (UCLP), and their perceptions of how their child was coping with treatment, comparing this information with recorded postsurgical complications.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 procedures for primary palatal repair were tested against a common procedure in the total cohort of 448 children born with a non-syndromic UCLP. A total of 356 parents completed the Scandcleft Parent Questionnaire, and 346 parents completed the Cleft Evaluation Profile.Results: The results indicated that the majority of parents were satisfied with cleft-related features of their child's appearance. Further, most children coped well with treatment according to their parents. Nevertheless, 17.5% of the children showed minor or short-term reactions after treatment experiences, and 2% had major or lasting difficulties. There were no significant relationships between parent perceptions of treatment-related problems and the occurrence of post-surgical medical complications.Conclusions: Most parents reported satisfaction with their child's appearance. However, treatment-related problems were described in some children, urging cleft centres to be aware of potential negative emotional and behavioural reactions to treatment in some young children, with a view to preventing the development of more severe treatment-related anxiety.Trial registration: ISRCTN29932826.

Background and aim: Normal articulation before school start is a main objective in cleft palate treatment. The aim was to investigate if differences exist in consonant proficiency at age 5 years between children with unilateral cleft lip and palate (UCLP) randomised to different surgical protocols 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, Norway, Sweden, 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 non-syndromic UCLP. Speech audio- and video-recordings of 391 children (136 girls and 255 boys) were available and transcribed phonetically. The main outcome measure was Percent Consonants Correct (PCC) from blinded assessments. Results: In Trial 1, arm A showed statistically significant higher PCC scores (82%) than arm B (78%) (p = .045). No significant differences were found between prevalences in Trial 2, A: 79%, C: 82%; or Trial 3, A: 80%, D: 85%. Across all trials, girls achieved better PCC scores, excluding s-errors, than boys (91.0% and 87.5%, respectively) (p = .01). Conclusions: PCC scores were higher in arm A than B in Trial 1, whereas no differences were found between arms in Trials 2 or 3. The burden of care in terms of secondary pharyngeal surgeries, number of fistulae, and speech therapy visits differed. Trial registration: ISRCTN29932826.

Normative language-based data are important for comparing speech performances of clinical groups. The Swedish Articulation and Nasality Test (SVANTE) was developed to enable a detailed speech assessment. This study's aim was to present normative data on articulation and nasality in Swedish speakers. Single word production, sentence repetition and connected speech were collected using SVANTE in 443 individuals. Mean (SD) and prevalences in the groups of 3-, 5-, 7-, 10-, 16- and 19-year-olds were calculated from phonetic transcriptions or ordinal rating. For the 3- and 5-year-olds, a consonant inventory was also determined. The mean percent of oral consonants correct ranged from 77% at age 3 to 99% at age 19. At age 5, a mean of 96% was already reached, and the consonant inventory was established except for /s/, /r/, /ɕ/. The norms on the SVANTE, also including a short version, will be useful in the interpretation of speech outcomes.

Mastering spatial and temporal co-ordination in speech production is a challenge for children. Voice onset time (VOT) reflects timing in speech. The objective was to study VOT in Swedish children with a diagnosed phonological impairment and compare results with normative data. Thus 38 children, aged 4-11 years, in three age-groups were audio-recorded when producing minimal pairs with the plosives /p b t d k g/. Waveforms and spectrograms were analysed. Results show that children with phonological impairment produced plosives with deviant VOT values and greater variability compared to normative data. No developmental trend was seen with increasing age. Also, no relationship was found between VOT values and degree of impairment measured by percentage phonemes correct. Furthermore no relation was found between number of errors on auditory discrimination of nine minimal pairs with the different plosives and number of deviant VOT. Findings were interpreted as displaying motor co-ordination difficulties.

Voice onset time (VOT) is a temporal acoustic parameter, which reflects the timing of speech motor control. The objective of the work was to obtain normative VOT data in Swedish children. Thus, 150 children aged 8-11 years old and 36 adults were audio-recorded when producing the plosives in minimal pairs. Measures were made using waveforms and spectro-grams. Results show that Swedish children developed adult-like VOT values between 9 and 10 years. By the age of 10 years prevoicing was also found to be completely adultlike in length. The results indicate that all Swedish adults do not produce voiced plosives with prevoicing. No evident gender differences were found. The obtained VOT values can be used as normative data when assessing children with speech and language disorders.

Objective. To evaluate outcome of two types of tonsil surgery (tonsillectomy [TE] + adenoidectomy or tonsillotomy [TT] + adenoidectomy) on vocal function perceptually and acoustically.Study Design. Sixty-seven children, aged 50-65 months, on waiting list for tonsil surgery were randomized to TE (n = 33) or TT (n = 34). Fifty-seven age-and gender-matched healthy preschool children were controls. Twenty-eight of them, aged 48-59 months, served as control group before surgery, and 29, aged 60-71 months, served as control group after surgery.Methods. Before surgery and 6 months postoperatively, the children were recorded producing three sustained vowels (/a/, /u/, and /i/) and 14 words. The control groups were recorded only once. Three trained speech and language pathologists performed the perceptual analysis using visual analog scale for eight voice quality parameters. Acoustic analysis from sustained vowels included average fundamental frequency, jitter percent, shimmer percent, noise-to-harmonic ratio, and the center frequencies of formants 1-3.Results. Before surgery, the children were rated to have more hyponasality and compressed/throaty voice (P < 0.05) and lower mean pitch (P < 0.01) in comparison to the control group. They also had higher perturbation measures and lower frequencies of the second and third formants. After surgery, there were no differences perceptually. Perturbation measures decreased but were still higher compared with those of control group (P < 0.05). Differences in formant frequencies for /i/ and /u/ remained. No differences were found between the two surgical methods.Conclusion. Voice quality is affected perceptually and acoustically by adenotonsillar hypertrophy. After surgery, the voice is perceptually normalized but acoustic differences remain. Outcome was equal for both surgical methods.

Tonsillar hypertrophy is common in young children and affects several aspects of the speech such as distortions of the dento-alveolar consonants. The study objective was to assess /s/-articulation, perceptually and acoustically, in children with tonsillar hypertrophy and compare effects of two types of surgery, total tonsillectomy and tonsillotomy. Sixty-seven children, aged 50-65 months, on the waiting list for surgery, were randomized to tonsillectomy or tonsillotomy. The speech material was collected preoperatively and 6 months postoperatively. Two groups of age-matched children were controls. /S/-articulation was affected acoustically with lower spectral peak locations and perceptually with less distinct /s/-production before surgery, in comparison to controls. After surgery /s/-articulation was normalized perceptually, but acoustic differences remained. No significant differences between surgical methods were found.

OBJECTIVES To compare two methods of tonsil surgery with respect to long term effect in a group of pre-school children METHODS 67 pre-school children, age 4-5 with symptoms of tonsillar hyperplasia were randomized to conventional tonsillectomy,TE,(n: 32) or tonsillotomy using radio frequency technique,TT(n:35). 28 TT patients and 25 TE patients underwent adenoidectomy at the same occasion. 5 TT and 1 TE had already undergone an adenoidectomy. Six months and two years after surgery all children were evaluated through questionnaires including the OSA-18 quality of life survey and two years after surgery they were again evaluated by an ENT-specialist, speech therapist and dentist. RESULTS The TT-children experienced less pain from the first day after surgery and were free from pain three days earlier than the TE-children. Six months after surgery there was no difference between the groups concerning snoring and frequency and severity of upper airway infections. After two years, three of the 35 TT-children and one of the TE children had been reoperated due to recurring obstructive problems, the TEchild and one of the TT-children with adenoidectomy, two TT-children with tonsillectomy. The otherchildren did not snore and no increased tendency towards infections was noted in any child. Evaluation with VAS before/two years after surgery was 8,4/1,3 for TE and 8,5/1,6 for TT.(Median). The total OSA-18 score and the scores for all domains within OSA 18 showed significant improvement after surgery for both groups of children(p

Adeno-tonsillar hypertrophy with obstructive sleep disordered breathing (OSDB) is known to affect oral-motor function, behaviour, and academic performance. Adeno-tonsillectomy is the most frequently performed operation in children, with total tonsillectomy (TE) being more common than partial resection, 'tonsillotomy' (TT). In the present study 67 children, aged 50-65 months, with OSBD were randomized to TE or TT. The children's phonology was assessed pre-operatively and 6 months post-operatively. Two groups of children served as controls. Phonology was affected in 62.7% of OSBD children before surgery, compared to 34% in the control group (p < .001). Also, OSBD children had more severe phonological deficits than the controls (p < .001). Phonology improved 6 months equally after both surgeries. Despite improvement post-operatively, the gap to the controls increased. Other functional aspects, such as oral motor function, were normalized regardless of surgical method--TE or TT. The impact of OSBD should be considered as one contributing factor in phonological impairment.

Objectives: Compare two techniques for pediatric tonsil surgery with respect to postoperative pain and morbidity and changes in sleep behavior, health related quality of life (HRQL) and benefits due to surgery.Methods: 67 children (4.5-5.5 years) with tonsillar hypertrophy and obstructive sleep-disordered breathing with or without recurrent tonsillitis were randomized to either regular tonsillectomy (TE) (n = 32) or intracapsular tonsillectomy/tonsillotomy (TT) (n = 35) with Radiofrequency surgical technique (ellman Int.). Before TT/TE, the parents completed a validated Quality of Life survey of pediatric obstructive sleep apnea, the OSA-18 (Obstructive Sleep Apnea-18) and a standardized assessment of their children's behavior with the Child Behavior Checklist (CBCL). Six months after surgery, the parents repeated these measurements, and assessed the health related benefits of the surgery using the Glasgow Children's Benefit Inventory (GCBI).Results: In the TT group, the children recorded less pain from the first day after surgery onwards, used fewer doses of painkillers and were pain-free 3 days earlier than the children in the TE group. Six months after surgery, there were no significant difference between TT and TE with regard to snoring and ENT-infections.The differences in the total scores and in all the individual domains between the initial OSA-18 and post-surgery scores were all significant (P < 0.0001). The improvement in the total problem score measured with CBCL was also significant (P < 0.01) and there was no difference between the TT and TE children. The improvements in all subscores of the GCBI indicated a significant health benefit of both TT and TE.Conclusions: TT with RF-surgery causes less pain and postoperative morbidity than regular TE and has an equal effect on snoring and recurrent infections.Pre-school children with tonsillar hypertrophy and obstructive sleep-disordered breathing all show an impact on HRQL and behavior before surgery and improve dramatically just as much after TT as after TE. Therefore TT would be considered for treatment of small children. (c) 2009 Elsevier Ireland Ltd. All rights reserved.

Adenotonsillar hypertrophy is associated with a wide range of problems. The enlargement causes obstructive symptoms and affects different functions such as chewing, swallowing, articulation, and voice. The objective of this study was to assess oral motor function in children with adenotonsillar hypertrophy using Nordic Orofacial Test-Screening (NOT-S) before and 6 months after surgery consisting of adenoidectomy combined with total or partial tonsil removal. A total of 67 children were assigned to either tonsillectomy (n=33) or partial tonsillectomy, 'tonsillotomy' (n=34); 76 controls were assessed with NOT-S and divided into a younger and older age group to match pre- and post-operated children. Most children in the study groups had oral motor problems prior to surgery including snoring, open mouth position, drooling, masticatory, and swallowing problems. Post-surgery oral motor function was equal to controls. Improvement was independent of surgery method.

Introduction: The objective of the present investigation was to compare two techniques for pediatric tonsil surgery with respect to postoperative pain and morbidity and changes in sleep behavior, health related quality of life (HRQL) and benefit due to surgery. Methods: 67 children (4,5-5,5 years) with tonsillar hypertrophy and obstructive sleep related distress with or without recurrent tonsillitis were randomized to either regular tonsillectomy (TE)(n=32) or intracapsular tonsillectomy/tonsillotomy (TT) (n=35) with Radiofrequency surgical technique (Ellman Int) Before TT/TE, the parents completed a validated Quality of Life survey of pediatric obstructive sleep apnea, the OSA18 (Obstructive Sleep Apnea-18) and a standardized assessment of their children-s behavior with the Child Behavior Checklist (CBCL). Six months after surgery, the parents repeated these measurements, and assessed the health related benefits of the surgery using the Glasgow Children´s Benefit Inventory (GCBI). Results: In the TT group, the children recorded less pain from the first day after surgery onwards, used fewer doses of painkillers and were pain-free 3 days earlier than the children in the TE group. Six months after surgery, there was no significant difference between TT and TE with regard to snoring and ENT-infections. The differences were all significant in the total scores and in all the individual domains between the initial OSA-18 and post-surgery scores (p<0.0001). The improvement in the total problem score measured with CBCL was also significant (p<0.01) and there were no differences between the TT and TE children. The improvements in all sub scores of the GCBI indicated a significant health benefit of both TT and TE. Conclusions: TT with RF-surgery is a safe method, which causes less pain and postoperative morbidity than regular TE and has a similar effect on snoring and recurrent infections. Young children with tonsillar hypertrophy and different degrees of obstructive sleep related distress all show an impact on HRQL and behavior. All improve dramatically after a tonsillar operation-improving just as much after TT as after TE. Based on these results, TT should be the first choice for treatment of these small children. Support: Financial support from the Research Council of South East Sweden (FORSS).