Mean GOSLON Yardstick Scores After 3 Different Treatment Protocols-A Long-term Study of Patients With Unilateral Cleft Lip and Palate.

OBJECTIVES: (1) To evaluate dental arch relationships, with the Great Ormond Street, London and Oslo (GOSLON) Yardstick, of participants with Unilateral cleft lip and palate (UCLP) and treated with 1-stage palatal closure with 3 different surgical protocols (2) to compare the mean GOSLON ratings with other CLP centers. DESIGN: Retrospective study of medical charts and dental models. SETTING: Karolinska University Hospital, Stockholm, Sweden. PARTICIPANTS: Eighty-seven patients with UCLP operated with 1-stage palatal repair. Thirty-five were operated with Veau-Wardill-Kilner (VWK) technique 1975 to 1986, 31 with minimal incision technique (MIT) from 1987 to 1997, and 21 according to MIT with muscle reconstruction (MITmr) 1998 to 2004. INTERVENTIONS: Dental casts at ages 5 (n = 87), 7 to 8 (n = 27), 10 (n = 81), 16 (n = 61), and 19 (n = 35) years were rated by 10 assessors with the GOSLON Yardstick. Information of other interventions was retrieved from patients' charts. MAIN OUTCOME MEASURES: Mean GOSLON ratings. RESULTS: A total of 82% of the participants were rated as having excellent to satisfactory outcome. Weighted κ statistics for the 10 assessors was good for inter-rater agreement and good/very good for intra-rater agreement. CONCLUSIONS: The mean GOSLON score in the Stockholm overall material at age 10 was 2.67. The VWK technique resulted in a greater need of orthognathic surgery than the MIT ( P < .01). The MITmr did not produce better dental arch relationships than MIT at age 5 ( P < .05). The best dental arch relationships were found in the MIT group at 10 years, mean 2.58, which is not significantly different from other centers with excellent outcome except Gothenburg and Vienna.

Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 4. Speech outcomes in 5-year-olds - velopharyngeal competency and hypernasality.

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.

Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 5. Speech outcomes in 5-year-olds - consonant proficiency and errors.

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.

A Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 1. Planning and management.

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.

Motor functions and adaptive behaviour in children with childhood apraxia of speech.

PURPOSE: Undiagnosed motor and behavioural problems have been reported for children with childhood apraxia of speech (CAS). This study aims to understand the extent of these problems by determining the profile of and relationships between speech/non-speech oral, manual and overall body motor functions and adaptive behaviours in CAS. METHOD: Eighteen children (five girls and 13 boys) with CAS, 4 years 4 months to 10 years 6 months old, participated in this study. The assessments used were the Verbal Motor Production Assessment for Children (VMPAC), Bruininks-Oseretsky Test of Motor Proficiency (BOT-2) and Adaptive Behaviour Assessment System (ABAS-II). RESULT: Median result of speech/non-speech oral motor function was between -1 and -2 SD of the mean VMPAC norms. For BOT-2 and ABAS-II, the median result was between the mean and -1 SD of test norms. However, on an individual level, many children had co-occurring difficulties (below -1 SD of the mean) in overall and manual motor functions and in adaptive behaviour, despite few correlations between sub-tests. CONCLUSION: In addition to the impaired speech motor output, children displayed heterogeneous motor problems suggesting the presence of a global motor deficit. The complex relationship between motor functions and behaviour may partly explain the undiagnosed developmental difficulties in CAS.

Speech results after one-stage palatoplasty with or without muscle reconstruction for isolated cleft palate.

OBJECTIVE: To investigate speech outcome between children with isolated cleft palate undergoing palatoplasty with and without muscle reconstruction and to compare speech outcomes between cleft and non cleft children. The number of subsequent velopharyngeal flaps was compared with respect to surgical techniques and cleft extent. DESIGN: Cross-sectional retrospective study. PARTICIPANTS: One hundred four children aged 4 years, 0 months to 6 years, 0 months, 33 with isolated cleft of the soft palate, 53 with isolated cleft of the hard and soft palate, and 18 non cleft children. INTERVENTIONS: Two primary palate repair techniques: minimal incision technique (MIT) and minimal incision technique including muscle reconstruction (MIT(mr)). MAIN OUTCOME MEASURES: Perceptual judgment of seven speech parameters assessed on a five-point scale. RESULTS: No significant differences in speech outcomes were found between MIT and MIT(mr) surgery groups. The number of velopharyngeal flaps was significantly lower after MIT(mr) surgery compared to MIT surgery. The number of flaps was also significantly lower in children with cleft of the soft palate compared to children with cleft of the hard and soft palate. Children with cleft of the soft palate had significantly less glottal articulation and weak pressure consonants compared to children with cleft of the hard and soft palate. CONCLUSIONS: The MIT(mr) surgery technique was not significantly superior to the MIT technique regarding speech outcomes related to velopharyngeal competence, but had fewer velopharyngeal flaps, which is contradictory. Until a larger sample can be studied, we will continue to use MIT(mr) for primary palate repair.

Universal parameters for reporting speech outcomes in individuals with cleft palate.

OBJECTIVE: To achieve consistency and uniformity in reporting speech outcomes in individuals born with cleft palate with or without cleft lip using perceptual parameters that characterize their speech production behavior regardless of the language or languages spoken. DESIGN: A working group of six individuals experienced in speech and cleft palate was formed to develop a system of universal parameters for reporting speech outcomes in individuals born with cleft palate. The system was adopted in conjunction with a workshop held in Washington, D.C., that was devoted to developing the universal system. The system, which was refined further following the workshop, involves a three-stage plan consisting of (1) evaluation, (2) mapping, and (3) reporting. The current report focuses primarily on the third stage, reporting speech outcomes. RESULTS: A set of five universal speech parameters has been devised for the reporting stage. These consist of (1) hypernasality, (2) hyponasality, (3) audible nasal air emission and/or nasal turbulence, (4) consonant production errors, and (5) voice disorder. Also included are speech understandability and speech acceptability, global parameters that can be reported for any type of speech disorder. The parameters are described in detail, and guidelines for speech-sampling content and scoring procedures in relation to the parameters are presented. CONCLUSION: A plan has been developed to document speech outcomes in individuals with cleft palate, regardless of the spoken language, using a set of five universal reporting parameters and two global speech parameters.

Speech outcome following treatment in cross-linguistic cleft palate studies: methodological implications.

OBJECTIVE: In cross-linguistic studies of cleft palate speech outcome following treatment, treatment is the independent variable, speech outcome is the dependent variable, and the speakers' language background is a background variable, like sex, age, and cleft type, which must be eliminated. This article focuses on language as a background variable and how it should be treated. The methodological problems are illustrated through a presentation of two cross-linguistic speech outcome studies. CONCLUSION: When speakers of different language background are included in cleft palate studies of speech outcome following treatment, speech outcome data should be based on speech units that are phonetically identical across languages. This affects the make-up of the speech material used in the study. In practice, the requirement of phonetically identical speech units may not be totally met, and detailed information regarding the interaction between the cleft condition and speech sound production is still required to fully understand how the validity of data is affected if this requirement is not met.

A four-year longitudinal study of palatal plate therapy in children with Down syndrome: effects on oral motor function, articulation and communication preferences.

The orofacial function in 20 children with Down syndrome was evaluated after 4 years of palatal plate therapy in 9 of the children (PPG); the remaining 11 were untreated age-matched controls (CG). All 20 children had received continuous orofacial physical therapy from their speech therapist during the treatment period. A clinical extra- and intraoral examination was performed, including oral motor function, facial expression, the occurrence of malocclusions, and hypertrophic tonsils. A questionnaire requesting data on breathing patterns, drooling, eating problems, and communicative preferences was answered by the parents. An articulation assessment was performed by two speech and language pathologists blinded to the treatment status of the children in order to find out whether the palatal plate had stimulated to improved oral speech behavior. The results for oral motor function showed significant differences between the groups in favor of the PPG for the summary variables for: visible tongue (P < 0.01), visible tongue during non-speech periods (P < 0.05), and lip-rounding during spontaneous speech (P < 0.01). During non-speech time, the PPG had their mouths open significantly less than the CG (P < 0.05). Expressivity of facial expression on a visual analog scale in the PPG scored 75.6 +/- 13.3 compared to 51.8 +/- 25.7 in the CG (P < 0.05). The intraoral examination showed that 6/9 children in the PPG and 7/11 in the CG had enlarged tonsils, resulting in more than 50% inter-tonsillary space reduction. Despite these findings, and no significant differences between the groups with respect to mouth/ nose breathing, nocturnal snoring was significantly less in the PPG than in the CG (P < 0.05), according to the parental questionnaire. After 4 years of palatal plate therapy, orofacial function had improved significantly in the 9 PPG children and specifically in terms of tongue position and lip activity.