Longitudinal Speech Outcome at 5 and 10 Years in UCLP: Influence of Speech Therapy and Secondary Velopharyngeal Surgery.

OBJECTIVE: To investigate speech development of children aged 5 and 10 years with repaired unilateral cleft lip and palate (UCLP) and identify speech characteristics when speech proficiency is not at 'peer level' at 10 years. Estimate how the number of speech therapy visits are related to speech proficiency at 10 years, and what factors are predictive of whether a child's speech proficiency at 10 years is at 'peer level' or not. DESIGN: Longitudinal complete datasets from the Scandcleft project. PARTICIPANTS: 320 children from nine cleft palate teams in five countries, operated on with one out of four surgical methods. INTERVENTIONS: Secondary velopharyngeal surgery (VP-surgery) and number of speech therapy visits (ST-visits), a proxy for speech intervention. MAIN OUTCOME MEASURES: 'Peer level' of percentage of consonants correct (PCC, > 91%) and the composite score of velopharyngeal competence (VPC-Sum, 0-1). RESULTS: Speech proficiency improved, with only 23% of the participants at 'peer level' at 5 years, compared to 56% at 10 years. A poorer PCC score was the most sensitive marker for the 44% below 'peer level' at 10-year-of-age. The best predictor of 'peer level' speech proficiency at 10 years was speech proficiency at 5 years. A high number of ST-visits received did not improve the probability of achieving 'peer level' speech, and many children seemed to have received excessive amounts of ST-visits without substantial improvement. CONCLUSIONS: It is important to strive for speech at 'peer level' before age 5. Criteria for speech therapy intervention and for methods used needs to be evidence-based.

Scandcleft randomized trials of primary surgery for unilateral cleft lip and palate: Speech proficiency at 10 years of age.

BACKGROUND & AIM: To assess consonant proficiency and velopharyngeal function in 10-year-old children born with unilateral cleft lip and palate (UCLP) within the Scandcleft project. METHODS & PROCEDURES: Three parallel group, randomized, clinical trials were undertaken as an international multicentre study by nine cleft teams in five countries. Three different surgical protocols for primary palate repair (Arm B-Lip and soft palate closure at 3-4 months, hard palate closure at 36 months, Arm C-Lip closure at 3-4 months, hard and soft palate closure at 12 months, and Arm D-Lip closure at 3-4 months combined with a single-layer closure of the hard palate using a vomer flap, soft palate closure at 12 months) were tested against a common procedure (Arm A-Lip and soft palate closure at 3-4 months followed by hard palate closure at 12 months) in the total cohort of 431 children born with a non-syndromic UCLP. Speech audio and video recordings of 399 children were available and perceptually analysed. Percentage of consonants correct (PCC) from a naming test, an overall rating of velopharyngeal competence (VPC) (VPC-Rate), and a composite measure (VPC-Sum) were reported. OUTCOMES & RESULTS: The mean levels of consonant proficiency (PCC score) in the trial arms were 86-92% and between 58% and 83% of the children had VPC (VPC-Sum). Only 50-73% of the participants had a consonant proficiency level with their peers. Girls performed better throughout. Long delay of the hard palate repair (Arm B) indicated lower PCC and simultaneous hard and soft palate closure higher (Arm C). However, the proportion of participants with primary VPC (not including velopharyngeal surgeries) was highest in Arm B (68%) and lowest in Arm C (47%). CONCLUSIONS & IMPLICATIONS: The speech outcome in terms of PCC and VPC was low across the trials. The different protocols had their pros and cons and there is no obvious evidence to recommend any of the protocols as superior. Aspects other than primary surgical method, such as time after velopharyngeal surgery, surgical experience, hearing level, language difficulties and speech therapy, need to be thoroughly reviewed for a better understanding of what has affected speech outcome at 10 years. WHAT THIS PAPER ADDS: What is already known on the subject Speech outcomes at 10 years of age in children treated for UCLP are sparse and contradictory. Previous studies have examined speech outcomes and the relationship with surgical intervention in 5-year-olds. What this study adds to the existing knowledge Speech outcomes based on standardized assessment in a large group of 10-year-old children born with UCLP and surgically treated according to different protocols are presented. While speech therapy had been provided, a large proportion of the children across treatment protocols still needed further speech therapy. What are the potential or actual clinical implications of this work? Aspects other than surgery and speech function might add to the understanding of what affects speech outcome. Effective speech therapy should be available for children in addition to primary surgical repair of the cleft and secondary surgeries if needed.

Observation method identifies that a lack of canonical babbling can indicate future speech and language problems.

AIM: We investigated how accurate observations of canonical babbling (CB) were and explored predictive babbling measures in children with and without medical diagnoses of conditions that can lead to speech and language problems. METHODS: From 2012 to 2014 this Stockholm-based study recruited 38 children aged nine months to 21 months with medical diagnoses and 30 children aged 10 months without diagnoses and included 21 previously studied 12-month-old children without medical diagnoses. CB and consonant sound production were directly observed by video recording natural play with a caregiver. The percentage of CB was calculated from each recording, and a validated observation form was used. How accurately the children with and without CB were classified was investigated with sensitivity and specificity. The groups were compared using predictive babbling variables. RESULTS: The observation method identified children with and without CB well, with a specificity of 0.89 and sensitivity of 0.93, respectively. Children with predictive babbling measures were identified in the clinical group (specificity 0.93-0.97), and a lack of these measures indicated a risk of being in the clinical group (odds ratios > 10). The sensitivity was low (0.32-0.42). CONCLUSION: Observation effectively identified a lack of CB and supported the importance of assessing babbling measures.

Speech problems affect more than one in two children with cerebral palsy: Swedish population-based study.

AIM: To describe speech ability in a population-based study of children with cerebral palsy (CP), in relation to CP subtype, motor function, cognitive level and neuroimaging findings. METHODS: A retrospective chart review of 129 children (66 girls, 63 boys) with CP, born in 1999-2002, was carried out. Speech ability and background information, such as type of CP, motor function, cognitive level and neuroimaging data, were collected and analysed. RESULTS: Speech disorders were found in 21% of the children and were present in all types of CP. Forty-one per cent of the children with speech disorders also had mental retardation, and 42% were able to walk independently. A further 32% of the children were nonverbal, and maldevelopment and basal ganglia lesions were most common in this group. The remaining 47% had no speech disorders, and this group was most likely to display white matter lesions of immaturity. CONCLUSION: More than half of the children in this CP cohort had a speech disorder (21%) or were nonverbal (32%). Speech ability was related to the type of CP, gross motor function, the presence of mental retardation and the localization of brain maldevelopment and lesions. Neuroimaging results differed between the three speech ability groups.

Exploring quantitative methods for evaluation of lip function.

The objective was to explore quantitative methods for the measurement of lip mobility and lip force and to relate these to qualitative assessments of lip function. Fifty healthy adults (mean age 45 years) and 23 adults with diagnoses affecting the facial muscles (mean age 37 years) participated in the study. Diagnoses were Möbius syndrome (n=5), Facioscapulohumeral muscular dystrophy (n=6) and Myotonic dystrophy type 1 (n=12). A system for computerised 3D analysis of lip mobility and a lip force meter were tested, and the results were related to results from qualitative assessments of lip mobility, speech (articulation), eating ability and saliva control. Facial expressions studied were open mouth smile and lip pucker. Normative data and cut-off values for adults on lip mobility and lip force were proposed, and the diagnostic value of these thresholds was tested. The proposed cut-off values could identify all inviduals with moderate or severe impairment of lip mobility but not always the milder cases. There were significant correlations between the results from quantitative measurements and qualitative assessments. The examined instruments for measuring lip function were found to be reliable with an acceptable measuring error. The combination of quantitative and qualitative ways to evaluate lip function made it possible to show the strong relation between lip contraction, lip force, eating ability and saliva control. The same combination of assessments can be used in the future to study if oral motor exercises aimed at improving lip mobility and strength could have a positive effect on lip function.

Peripheral facial palsy: Speech, communication and oral motor function.

OBJECTIVES: The aim of the present study was to examine the effect of acquired unilateral peripheral facial palsy on speech, communication and oral functions and to study the relationship between the degree of facial palsy and articulation, saliva control, eating ability and lip force. MATERIALS AND METHODS: In this descriptive study, 27 patients (15 men and 12 women, mean age 48years) with unilateral peripheral facial palsy were included if they were graded under 70 on the Sunnybrook Facial Grading System. The assessment was carried out in connection with customary visits to the ENT Clinic and comprised lip force, articulation and intelligibility, together with perceived ability to communicate and ability to eat and control saliva conducted through self-response questionnaires. RESULTS: The patients with unilateral facial palsy had significantly lower lip force, poorer articulation and ability to eat and control saliva compared with reference data in healthy populations. The degree of facial palsy correlated significantly with lip force but not with articulation, intelligibility, perceived communication ability or reported ability to eat and control saliva. CONCLUSION: Acquired peripheral facial palsy may affect communication and the ability to eat and control saliva. Physicians should be aware that there is no direct correlation between the degree of facial palsy and the possible effect on communication, eating ability and saliva control. Physicians are therefore recommended to ask specific questions relating to problems with these functions during customary medical visits and offer possible intervention by a speech-language pathologist or a physiotherapist.

ABR thresholds in infants born with CLP and OME and infants with OME.

OBJECTIVES: The aim of this study was to investigate and compare auditory brainstem response (ABR) thresholds related to otitis media with effusion (OME) in infants with and without cleft palate and/or lip (CP±L). METHODS: Forty-seven infants with CP±L and 67 infants with OME participated in the study. Hearing thresholds of ears of infants with OME were compared between groups and within the group with CP±L. RESULTS: Infants with CP±L and OME presented with similar hearing thresholds as infants with OME and not CP±L. Within the cleft group, infants with isolated cleft palate and OME demonstrated significantly higher hearing thresholds than infants with unilateral cleft lip and palate and OME. CONCLUSION: A high prevalence of infants with CP±L present with OME early in life. Hearing thresholds of these infants are similar to infants without CP±L, but with OME. The ear status and hearing thresholds of infants with CP±L needs to be monitored to be able to provide the best access to hearing in order to fully allow speech and language development.

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.

NORAM: calibration and operational advice for measuring nasality in cleft palate patients.

This study was carried out to evaluate the NORAM (Nasal-Oral-RAtio-Meter), developed at the Department of Speech Communication and Music Acoustics, RIT. The speech samples used were the recorded speech of normal and nasally deviant speakers. NORAM measures the total speech time and the duration of the nasalized portions, it also calculates the ratio between these two values. The signals are picked up by two contact microphones, one placed on the alar cartilage of the nose and the other on the outside of the lamina of the thyroid cartilage. They are compared and the segment is rated as nasal, if the signal from the nose pick-up is close in intensity to the larynx signal. It was found that the threshold should be set 9 dB below the larynx signal. The reliability of the measurements depends to a large degree on the accurate calibration of the instrument. In sentences lacking nasal consonants, produced by normal speakers, some nasal segments were registered at word boundaries and at the end of phrases. In the patient material these segments tended to be broader and additional segments were also found.