Elisabeth Willadsen

Methodology for Speech Assessment in the Scandcleft Project—An International Randomized Clinical Trial on Palatal Surgery: Experiences from a Pilot Study:

Objective: To present the methodology for speech assessment in the Scandcleft project and discuss issues from a pilot study. Design: Description of methodology and blinded test for speech assessment. Speech samples and instructions for data collection and analysis for comparisons of speech outcomes across five included languages were developed and tested. Participants and Materials: Randomly selected video recordings of 10 5-year-old children from each language (n  =  50) were included in the project. Speech material consisted of test consonants in single words, connected speech, and syllable chains with nasal consonants. Five experienced speech and language pathologists participated as observers. Main Outcome Measures: Narrow phonetic transcription of test consonants translated into cleft speech characteristics, ordinal scale rating of resonance, and perceived velopharyngeal closure (VPC). A velopharyngeal composite score (VPC-sum) was extrapolated from raw data. Intra-agreement comparisons were performed. Results: Range for intra-agreement for consonant analysis was 53% to 89%, for hypernasality on high vowels in single words the range was 20% to 80%, and the agreement between the VPC-sum and the overall rating of VPC was 78%. Conclusions: Pooling data of speakers of different languages in the same trial and comparing speech outcome across trials seems possible if the assessment of speech concerns consonants and is confined to speech units that are phonetically similar across languages. Agreed conventions and rules are important. A composite variable for perceptual assessment of velopharyngeal function during speech seems usable; whereas, the method for hypernasality evaluation requires further testing.

Influence of Timing of Hard Palate Repair in a Two-Stage Procedure on Early Speech Development in Danish Children With Cleft Palate

Objective To investigate the influence of timing of hard palate closure on early speech development from 18 months to 3 years of age. Design A prospective, randomized clinical trial. Participants Thirty-four children with unilateral cleft lip and palate (UCLP) with velum closure at 4 months of age, and hard palate closure at 12 months (early hard palate repair, EarlyHPR) or 36 months (late hard palate unrepaired, LateHPU) by random assignment. Thirty-five control children were matched for gender and age. Methods All children were video recorded during a play interaction with a parent at 18 months of age. These recordings were transcribed according to the International Phonetic Alphabet. At 36 months a single word naming test was administered. Results At 18 months the LateHPU group produced fewer labial stops and more velar stops than the EarlyHPR group. Unlike the EarlyHPR group, the LateHPU group produced fewer vocalizations, consonants, and consonants permissible in word-initial position than the control group. Additionally, both cleft palate groups had a smaller productive vocabulary than the control group, but unlike the EarlyHPR group, the LateHPU group produced a smaller number of word tokens in social interaction than the control group. By 3 years of age, the LateHPU group had a (severely) restricted phonological system and produced more cleft speech characteristics than the EarlyHPR group. Conclusions Surgical timing of hard palate repair in a two-stage procedure appears to have an influence on early speech development in children with cleft palate.

Phonetic Description of Babbling in Danish Toddlers Born With and Without Unilateral Cleft Lip and Palate

Objective To investigate prelinguistic vocalization sequences of 1-year-old children with and without cleft lip and palate. Design Prospective study. Participants Thirty-eight children born with unilateral cleft lip and palate and 36 control children born without clefts. The cleft children had the lip, soft palate, and posterior part of the hard palate repaired at 4 months of age. The lip was closed ad modum Millard, the nose was corrected according to McComb, and the soft palate was closed with a posteriorly based vomer flap. Methods Data were obtained from a clinical visit during which the baby played with the mother. Video recordings were transcribed and analyzed concerning (1) the frequency of occurrence of vocalization sequences, (2) the frequency of occurrence of contoids and vocoids, (3) the contoids’ place and manner of articulation, and (4) the percentage of children who entered the canonical babbling stage. Results No significant differences were observed between the cleft and control groups concerning frequency of occurrence of vocalization sequences, contoids, or vocoids. Structural differences between the groups seem to influence the contoid inventory, with a higher frequency of occurrence of nasal contoids and a smaller frequency of occurrence of alveolar contoids in the cleft group. Canonical babbling was achieved by most children in both groups, and no significant difference was found between the groups. Conclusions Early closure of the soft palate seems to have a positive influence on the prelinguistic development of children with cleft palate.

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 comparative study of prespeech vocalizations in two groups of toddlers with cleft palate and a noncleft group.

OBJECTIVE This study examined the prelinguistic contoid (consonant-like) inventories of 14 children with unilateral cleft lip and palate (C-UCLP) at 13 months of age. The children had received primary veloplasty at 7 months of age and closure of the hard palate was performed at 3-5 years. The results of this investigation were compared to results previously reported for 19 children with cleft palate and 19 noncleft children at the age of 13 months. The children with clefts in that study received a two-stage palatal surgery. This surgical procedure was formerly used at our center and included closure of the lip and hard palate at 3 months of age and soft palate closure at 22 months of age. DESIGN Retrospective study. SETTING The participants were videorecorded in their homes during play with their mothers. The videotapes were transcribed independently by three trained speech pathologists. PATIENTS Fourteen consecutive patients born with C-UCLP and no known mental retardation or associated syndromes served as subjects. RESULTS The children who received delayed closure of the hard palate demonstrated a significantly richer variety of contoids in their prespeech vocalizations than the cleft children in the comparison group. Both groups of subjects with clefts had significantly fewer plosives in their contoid inventory than the noncleft group, and there was no difference regarding place of articulation between the group that received delayed closure of the hard palate and the noncleft group.

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

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.

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

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. 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

Abstract 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 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 childrens 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.

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

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 < 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.