Timing of Primary Surgery for Cleft Palate.

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.).

Inter-rater reliability in classification of canonical babbling status based on canonical babbling ratio in infants with isolated cleft palate randomised to Timing of Primary Surgery for Cleft Palate (TOPS).

Canonical babbling (CB) is commonly defined as present when at least 15% of all syllables produced are canonical, in other words a canonical babbling ratio (CBR) ≥0.15. However, there is limited knowledge about inter-rater reliability in classification of CB status based on CBR and inter-rater differences in assessment of CBR. We investigated inter-rater reliability of experienced Speech Language Therapists (SLTs) on: classification of CB status based on CBR ≥ 0.15, CBRs and the total number of syllables per infant used to calculate CBR.Each infant (n = 484) was video-recorded at a clinical site in play interaction with their parent as part of the randomised controlled trial Timing of Primary Surgery for Cleft Palate. Each recording was subsequently assessed by three independent SLTs, from a pool of 29 SLTs. They assessed the recordings in real time.The three assessing SLTs agreed in classification of CB status in 423 (87.4%) infants, with higher complete agreement for canonical (91%; 326/358) than non-canonical (77%; 97/126). The average difference in CBR and total number of syllables identified between the SLT assessments of each infant was 0.12 and 95, respectively.This study provided new evidence that one trained SLT can reliably classify CB status (CBR ≥ 0.15) in real time when there is clear distinction between the observed CBR and the boundary (0.15); however, when the observed CBR approaches the boundary multiple SLT assessments are beneficial. Thus, we recommend to include assessment of inter-rater reliability, if the purpose is to compare CBR and total syllable count across infants or studies.Trial registration number here: www.clinicaltrials.gov, identifier NCT00993551.

A randomised controlled trial comparing palate surgery at 6 months versus 12 months of age (the TOPS trial): a statistical analysis plan.

BACKGROUND: 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 12 months (corrected for gestational age). METHODS/DESIGN: The TOPS trial is an international, two-arm, parallel group, randomised controlled trial. The primary outcome is insufficient velopharyngeal function at 5 years of age. Secondary outcomes, measured at 12 months, 3 years, and 5 years of age, include measures of speech 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. DISCUSSION: 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. TRIAL REGISTRATION: ClinicalTrials.gov NCT00993551 . Registered on 9 October 2009.

Adding a fourth rater to three had little impact in pre-linguistic outcome classification.

The consequence of differing levels of agreement across raters is rarely studied. Subsequently, knowledge is limited on how number of raters affects the outcome. The present study aimed to examine the impact on pre-linguistic outcome classifications of 12-month-old infants when using four raters compared to three. Thirty experienced Speech and Language Therapists (SLTs) from five countries assessed 20 minute video recordings of four 12-month-old infants during a play session with a parent. One recording was assessed twice. A naturalistic listening method in real time was used. This involved: (1) assessing, each syllable as canonical or non-canonical, and (2) following the recording, assessing if the infant was babbling canonically and listing the syllables the infant produced with command. The impact that four raters had on outcome, compared to three, was explored by classifying the outcome based on all possible combinations of three raters and determining the frequency that the outcome assessment changed when a fourth assessor was added. Results revealed that adding a fourth rater had a minimal impact on canonical babbling ratio assessment. Presence/absence of canonical babbling and size of consonant inventory showed a negligible impact on three out of four recordings, whereas the size of syllable inventory and presence/absence of canonical babbling was minimally affected in one recording by adding a fourth rater. In conclusion, adding a forth rater in assessment of pre-linguistic utterances in 12-month-old infants with naturalistic assessment in real time does not affect outcome classifications considerably. Thus, using three raters, as opposed to four, is recommended.

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.

OBJECTIVE: 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. DESIGN: A prospective randomized clinical trial. SETTING: Two Norwegian and 2 British centers. PARTICIPANTS: 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). MAIN OUTCOME MEASURES: 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). RESULTS: 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). CONCLUSIONS: 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.

Longitudinal study of the development of obstruent correctness from ages 3 to 5 years in 108 Danish children with unilateral cleft lip and palate: a sub-study within a multicentre randomized controlled trial.

BACKGROUND: Speech-sound development in preschoolers with unilateral cleft lip and palate (UCLP) as a group is delayed/disordered, and obstruents comprise the most vulnerable sound class. AIMS: To evaluate the development of obstruent correctness (PCC-obs) and error types (cleft speech characteristics (CSCs) and developmental speech characteristics (DSCs)) from ages 3-5 and to investigate possible predictors (error types, velopharyngeal dysfunction (VPD) and gender) of PCC-obs at age 5 in two groups of children with UCLP. METHODS & PROCEDURES: Subgroup analysis was conducted within a multicentre randomized controlled trial (RCT) of primary surgery (Scandcleft Project). A total of 125 Danish children with UCLP received lip and soft palate repair around 4 months of age and early hard palate closure at 12 months (EHPC group) or late hard palate closure at 36 months (LHPC group). Audio and video recordings of a naming test were available for 108 children at ages 3 and 5, and recordings were transcribed phonetically by blinded raters. OUTCOMES & RESULTS: PCC-obs scores increased significantly from ages 3-5 in both groups, but with small effect sizes in the EHPC group that had higher scores at age 3 than the LHPC group. DSCs decreased in both groups whereas CSCs only decreased in the LHPC group that had more CSCs at age 3 than the EHPC group. The frequency of CSCs at age 3 was a significant predictor of PCC-obs scores at age 5 in both groups. DSCs significantly improved the logistic regression model in the EHPC group, whereas VPD and gender did not significantly improve the model in either group. CONCLUSIONS & IMPLICATIONS: Although PCC-obs developed significantly from ages 3 to 5, children with UCLP as a group did not catch up to typically developing Danish children at age 5. Furthermore, the LHPC group at age 5 did not reach the 3-year level of the EHPC group, which means that delaying hard palate closure until age 3 is detrimental to obstruent development. Both CSCs and DSCs at age 3 were important predictors of PCC-obs at age 5 and should be considered when determining need for intervention.

Assessment of prelinguistic vocalizations in real time: a comparison with phonetic transcription and assessment of inter-coder-reliability.

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.

Scandcleft Project Trial 2-Comparison of Speech Outcome in 1- and 2-Stage Palatal Closure in 5-Year-Olds With UCLP.

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.

Influence of Infant Cleft Dimensions on Velopharyngeal Function in 5-Year-Old Danish Children Born With Unilateral Cleft Lip and Palate.

AIM: To examine the association of cleft severity at infancy and velopharyngeal competence in preschool children with unilateral cleft lip and palate operated with early or delayed hard palate repair. DESIGN: Subgroup analysis within a multicenter randomized controlled trial of primary surgery (Scandcleft). SETTING: Tertiary health care. One surgical center. PATIENTS AND METHODS: One hundred twenty-five infants received cheilo-rhinoplasty and soft palate repair at age 3 to 4 months and were randomized to hard palate closure at age 12 or 36 months. Cleft size and cleft morphology were measured 3 dimensionally on digital models, obtained by laser surface scanning of preoperative plaster models (mean age: 1.8 months). MAIN OUTCOME MEASUREMENTS: Velopharyngeal competence (VPC) and hypernasality assessed from a naming test (VPC-Sum) and connected speech (VPC-Rate). In both scales, higher scores indicated a more severe velopharyngeal insufficiency. RESULTS: No difference between surgical groups was shown. A low positive correlation was found between posterior cleft width and VPC-Rate (Spearman = .23; P = .025). The role of the covariate "cleft size at tuberosity level" was confirmed in an ordinal logistic regression model (odds ratio [OR] = 1.17; 95% confidence interval [CI]:1.01-1.35). A low negative correlation was shown between anteroposterior palatal length and VPC-Sum (Spearman = -.27; P = .004) and confirmed by the pooled scores VPC-Pooled (OR = 0.82; 95% CI: 0.69-0.98) and VPC-Dichotomic (OR = 0.82; 95% CI: 0.68-0.99). CONCLUSIONS: Posterior cleft dimensions can be a modest indicator for the prognosis of velopharyngeal function at age 5 years, when the soft palate is closed first, independently on the timing of hard palate repair. Antero-posterior palatal length seems to protect from velopharyngeal insufficiency and hypernasality. However, the association found was significant but low.

Timing Of Primary Surgery for cleft palate (TOPS): protocol for a randomised trial of palate surgery at 6 months versus 12 months of age.

INTRODUCTION: 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, speech, hearing, dental development and facial growth. Over recent decades, age at palatal surgery in infancy has reduced. This has led to palatal closure in one-stage procedures being carried out around the age of 12 months, but in some cases as early as 6 months. The primary objective of the Timing Of Primary Surgery for Cleft Palate (TOPS)trial is to determine whether surgery for cleft palate performed at 6 or 12 months of age is most beneficial for speech outcomes. METHODS AND ANALYSIS: Infants with a diagnosis of non-syndromic isolated cleft palate will be randomised to receive standardised primary surgery (Sommerlad technique) for closure of the cleft at either 6 months or 12 months, corrected for gestational age. The primary outcome will be perceived insufficient velopharyngeal function at 5 years of age. Secondary outcomes measured across 12 months, 3 years and 5 years will include growth, safety of the procedure, dentofacial development, speech, hearing level and middle ear function. Video and audio recordings of speech will be collected in a standardised age-appropriate manner and analysed independently by multiple speech and language therapists. The trial aims to recruit and follow-up 300 participants per arm. Data will be analysed according to the intention-to-treat principle using a 5% significance level. All analyses will be prespecified within a full and detailed statistical analysis plan. ETHICS AND DISSEMINATION: Ethical approval has been sought in each participating country according to country-specific procedures. Trial results will be presented at conferences, published in peer-reviewed journals and disseminated through relevant patient support groups. TRIAL REGISTRATION NUMBER: NCT00993551; Pre-results.

Scandcleft Project, Trial 1: Comparison of Speech Outcome in Relation to Timing of Hard Palate Closure in 5-Year-Olds With UCLP.

OBJECTIVE: To compare in-depth speech results in Scandcleft Trial 1 as well as reference data from peers without cleft palate (CP). DESIGN: A prospective randomized clinical trial. SETTING: A Danish and a Swedish CP center. PARTICIPANTS: 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). MAIN OUTCOME MEASURES: 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. RESULTS: 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. CONCLUSIONS: 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.

A software program to assist coding of prelinguistic vocalizations in real time.

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.

Influence of timing of delayed hard palate closure on articulation skills in 3-year-old Danish children with unilateral cleft lip and palate.

BACKGROUND: Differing results regarding articulation skills in young children with cleft palate (CP) have been reported and often interpreted as a consequence of different surgical protocols. AIMS: To assess the influence of different timing of hard palate closure in a two-stage procedure on articulation skills in 3-year-olds born with unilateral cleft lip and palate (UCLP). Secondary aims were to compare results with peers without CP, and to investigate if there are gender differences in articulation skills. Furthermore, burden of treatment was to be estimated in terms of secondary surgery, hearing and speech therapy. METHODS & PROCEDURES: A randomized controlled trial (RCT). Early hard palate closure (EHPC) at 12 months versus late hard palate closure (LHPC) at 36 months in a two-stage procedure was tested in a cohort of 126 Danish-speaking children born with non-syndromic UCLP. All participants had the lip and soft palate closed around 4 months of age. Audio and video recordings of a naming test were available from 113 children (32 girls and 81 boys) and were transcribed phonetically. Recordings were obtained prior to hard palate closure in the LHPC group. The main outcome measures were percentage consonants correct adjusted (PCC-A) and consonant errors from blinded assessments. Results from 36 Danish-speaking children without CP obtained previously by Willadsen in 2012 were used for comparison. OUTCOMES & RESULTS: Children with EHPC produced significantly more target consonants correctly (83%) than children with LHPC (48%; p < .001). In addition, children with LHPC produced significantly more active cleft speech characteristics than children with EHPC (p < .001). Boys achieved significantly lower PCC-A scores than girls (p = .04) and produced significantly more consonant errors than girls (p = .02). No significant differences were found between groups regarding burden of treatment. The control group performed significantly better than the EHPC and LHPC groups on all compared variables.

Development and validation of a screening procedure to identify speech-language delay in toddlers with cleft palate.

The purpose of this study was to develop and validate a clinically useful speech-language screening procedure for young children with cleft palate ± cleft lip (CP) to identify those in need of speech-language intervention. Twenty-two children with CP were assigned to a +/- need for intervention conditions based on assessment of consonant inventory using a real-time listening procedure in combination with parent-reported expressive vocabulary. These measures allowed evaluation of early speech-language skills found to correlate significantly with later speech-language performance in longitudinal studies of children with CP. The external validity of this screening procedure was evaluated by comparing the +/- need for intervention assignment determined by the screening procedure to experienced speech-language pathologist (SLP)s' clinical judgement of whether or not a child needed early intervention. The results of real-time listening assessment showed good-excellent inter-rater agreement on different consonant inventory measures. Furthermore, there was almost perfect agreement between the children selected for intervention with the screening procedure and the clinical judgement of experienced SLPs indicate that the screening procedure is a valid way of identifying children with CP who need early intervention.

Validity of auditory perceptual assessment of velopharyngeal function and dysfunction - the VPC-Sum and the VPC-Rate.

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.

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.

Lexical selectivity in danish toddlers with cleft palate.

Objective : To study if Danish children with cleft palate display lexical selectivity in their early lexicon at 18 months of age. Design : A cross-sectional study. Participants : Thirty-four children with unilateral cleft lip and palate and 35 children without cleft palate, matched for gender and age. Methods : All participants were video recorded at 18 months of age during play interaction with a parent. The video recordings were transcribed according to the International Phonetic Alphabet and an individual consonant inventory was established for each participant. The video recordings were also analyzed with respect to word productions, establishing an observed productive vocabulary size for each participant. Results : At 18 months of age Danish children with cleft palate showed marked lexical selectivity in their early words. The distribution of consonant classes observed at 11 months of age in a previous study of the children with cleft palate was almost perfectly reflected in their early lexicon at 18 months. The early lexicon of children with cleft palate differed from the early lexicon of their noncleft peers. Conclusions and Implications : Danish toddlers with cleft palate display lexical selectivity in the early lexicon as it has been described for English-speaking toddlers with and without cleft palate, even though some qualitative differences were found.

A restricted test of single-word intelligibility in 3-year-old children with and without cleft palate.

OBJECTIVE: In a previous study, children with cleft palate with hard palate closure at 12 months of age showed more typical phonological development than did children with an unrepaired hard palate at 36 months of age. This finding was based on narrow transcription of word initial target consonants obtained from a simple naming test. To evaluate the relevance of this finding, we investigated how well the children's target words were understood by 84 naive listeners. DESIGN: A cross-sectional study. PARTICIPANTS: Data obtained from 28 children with unilateral cleft lip and palate, 3 years of age, who received hard palate closure at either 12 months (hard palate repaired; HPR) or 36 months (hard palate unrepaired; HPU) were compared with data obtained from 14 age-matched, typically developing control children. METHODS: Video recordings of the children naming target words were shown to 84 naive listeners between 15 and 24 years of age who typed the word they heard. RESULTS: The findings of this study indicated that naive listeners correctly identified a larger percentage of words in the control children followed by children in the HPR group. Children in the HPU group were more difficult for the naive listeners to understand. The error of retraction/backing of alveolar target consonants to velar place of articulation occurred frequently and most often in the HPU group and was found to have a negative effect on intelligibility.