Achieving the next level in cleft speech intervention: A protocol of a randomized sham-controlled trial to provide guidelines for a personalized approach in children with cleft palate.

BACKGROUND: Compensatory cleft speech disorders can severely impact speech understandability and speech acceptability. Speech intervention is necessary to eliminate these disorders. There is, however, currently no consensus on the most effective speech therapy approach to eliminate the different subtypes of compensatory cleft speech disorders. AIMS: To compare the immediate, short- and long-term effects of three well-defined speech intervention approaches (i.e., a motor-phonetic approach, a linguistic-phonological approach and a combined phonetic-phonological approach) on the speech and health-related quality of life (HRQoL) in Belgian Dutch-speaking children with cleft palate with or without cleft lip (CP±L) and different subtypes of compensatory speech disorders (i.e., anterior oral cleft speech characteristics (CSCs), posterior oral CSCs or non-oral CSCs). Besides, the perceived acceptability of these three speech intervention approaches will be investigated from the perspectives of caregivers and children with a CP±L. METHODS & PROCEDURES: A two-centre longitudinal randomized sham-controlled trial was used. Children were randomly assigned to one of the three intervention programmes and received 10 h of speech intervention divided over 2 weeks. Block randomization was used, stratified by age and gender. Primary outcome measures included perceptual speech outcomes. Secondary outcome measures included patient-reported outcomes. OUTCOMES & RESULTS: The results of this trial will provide speech-language pathologists evidence-based guidelines to better tailor intervention approaches to the specific needs of a child with a defined compensatory speech disorder. WHAT THIS PAPER ADDS: What is already known on this subject Speech therapy approaches to address cleft palate speech disorders are broadly divided into two categories: motor-phonetic interventions and linguistic-phonological interventions. Some limited evidence demonstrated the positive effects of these approaches in eliminating compensatory cleft speech disorders. Different studies have reported inter-individual variation, suggesting that one child may benefit more from a particular intervention approach than the other child. Perhaps this variation can be attributed to the specific subtype of compensatory speech disorder (i.e., anterior oral CSC, posterior oral CSC or non-oral CSC). What this paper adds to existing knowledge This paper describes a randomized sham-controlled trial that compared the immediate, short- and long-term effects of three well-defined speech intervention approaches (i.e., a motor-phonetic approach, a linguistic-phonological approach and a combined phonetic-phonological approach) on the speech and HRQoL in Belgian Dutch-speaking children with CP±L and different subtypes of compensatory cleft speech disorders (i.e., anterior oral CSCs, posterior oral CSCs or non-oral CSCs) measured by perceptual and psychosocial outcome measures. Besides, the experienced acceptability of these three speech intervention approaches were investigated from the perspectives of caregivers and children. What are the potential or actual clinical implications of this work? This project provides evidence-based knowledge on patient-tailored cleft speech intervention considering both scientific evidence and the perspectives of caregivers and children. The results aid SLPs in better tailoring intervention approaches to the needs of a child with a specific type of compensatory cleft speech disorder.

Speech outcomes after palatal closure in 3-7-year-old children.

INTRODUCTION: One of the main goals of the team approach in management of oro-facial clefts is to help the children with cleft palate have adequate speech development. OBJECTIVE: The present study aimed to investigate the prevalence of articulation and resonance disorders following palate closure in children who were visited for routine examination by the Isfahan Cleft Care Team between 2011 and 2015, and to study the impact of cleft type and age at the time of palatoplasty on speech outcomes. METHODS: Clinical records of 180 preschool children with repaired cleft palate were reviewed. The percentage of children demonstrating hypernasality, nasal emission, nasal turbulence, and compensatory misarticulations was calculated. The relationship between cleft type and age at the time of palatal surgery, as independent variables, and speech outcomes were examined. RESULTS: 67.7 and 64.5 percent of the children demonstrated respectively moderate/severe hypernasality and nasal emission, and 71.1 percent produced compensatory misarticulations. Age at the time of palatal repair was significantly associated with compensatory misarticulations and also with moderate/severe hypernasality. The prevalence of compensatory misarticulations, significant hypernasality, nasal emission and also nasal turbulence was not significantly different in various types of cleft. CONCLUSIONS: We observed a high prevalence of different speech disorders in preschool children with repaired cleft palate compared to other studies. This can be partly due to late palatal repair in the studied population. Despite many advances in cleft palate management programs in Iran, there are still many children who do not access the interdisciplinary team cares in their early childhood. We should, therefore, try to increase accessibility of appropriate and timely management services to all Iranian children with cleft lip/palate.

Brief Report: "Um" Fillers Distinguish Children With and Without ASD.

Two laboratories have reported that children with ASD are less likely than their typical peers to fill pauses with um but their use of uh is unaffected (Irvine et al., J Autism Dev Disord 46(3):1061-1070, 2016; Gorman et al., Autism Res 9(8):854-865, 2016). In this brief report, we replicated this finding by comparing the discourse of 7-to-15-year-olds with ASD (N = 31) to that of their typically developing same-age peers (N = 32). The robustness of this easily documented difference in discourse suggests a potentially useful clinical marker of ASD.

Classification of Articulator Movements and Movement Direction from Sensorimotor Cortex Activity.

For people suffering from severe paralysis, communication can be difficult or nearly impossible. Technology systems called brain-computer interfaces (BCIs) are being developed to assist these people with communication by using their brain activity to control a computer without any muscle activity. To benefit the development of BCIs that employ neural activity related to speech, we investigated if neural activity patterns related to different articulator movements can be distinguished from each other. We recorded with electrocorticography (ECoG), the neural activity related to different articulator movements in 4 epilepsy patients and classified which articulator participants moved based on the sensorimotor cortex activity patterns. The same was done for different movement directions of a single articulator, the tongue. In both experiments highly accurate classification was obtained, on average 92% for different articulators and 85% for different tongue directions. Furthermore, the data show that only a small part of the sensorimotor cortex is needed for classification (ca. 1 cm2). We show that recordings from small parts of the sensorimotor cortex contain information about different articulator movements which might be used for BCI control. Our results are of interest for BCI systems that aim to decode neural activity related to (actual or attempted) movements from a contained cortical area.

Use of electropalatography in the treatment of speech disorders in children with Down syndrome: a randomized controlled trial.

BACKGROUND: Electropalatography (EPG) records details of the location and timing of tongue contacts with the hard palate during speech. It has been effective in treating articulation disorders that have failed to respond to conventional therapy approaches but, until now, its use with children and adolescents with intellectual/learning disabilities and speech disorders has been limited. AIMS: To evaluate the usefulness of EPG in the treatment of speech production difficulties in children and adolescents with Down syndrome (DS) aged 8-18 years. METHODS & PROCEDURES: A total of 27 children with DS were assessed on a range of cognitive and speech and language measures and underwent additional EPG assessment. Participants were randomly allocated to one of three age-matched groups receiving either EPG therapy, EPG-informed conventional therapy or 'treatment as usual' over a 12-week period. The speech of all children was assessed before therapy using the Diagnostic Evaluation of Articulation and Phonology (DEAP) and reassessed immediately post- and 3 and 6 months post-intervention to measure percentage consonants correct (PCC). EPG recordings were made of the DEAP assessment items at all time points. Per cent intelligibility was also calculated using the Children's Speech Intelligibility Measure (CSIM). OUTCOMES & RESULTS: Gains in accuracy of production immediately post-therapy, as measured by PCC, were seen for all groups. Reassessment at 3 and 6 months post-therapy revealed that those who had received therapy based directly on EPG visual feedback were more likely to maintain and improve on these gains compared with the other groups. Statistical testing showed significant differences between groups in DEAP scores across time points, although the majority did not survive post-hoc evaluation. Intelligibility across time points, as measured by CSIM, was also highly variable within and between the three groups, but despite significant correlations between DEAP and CSIM at all time points, no statistically significant group differences emerged. CONCLUSIONS & IMPLICATIONS: EPG was an effective intervention tool for improving speech production in many participants. This may be because it capitalizes on the relative strength of visual over auditory processing in this client group. The findings would seem to warrant an increased focus on addressing speech production difficulties in current therapy.

Electrophysiological basis of reading related phonological impairment in Chinese speakers with schizophrenia: An ERP study.

It has been reported in alphabetic languages that individuals with schizophrenia showed language-related cognitive impairments including phonological deficits, which were in turn associated with clinical symptoms such as auditory hallucinations and thought disorders. To date, however, the phonological deficits involved in schizophrenia in Chinese and its neural basis have not been well established. In order to establish such a relationship we conducted a behavioral study using lexical tone judgment and digit span tasks as well as an event-related potential (ERP) study with an auditory oddball paradigm, in particular, for P300 effects, the event-related brain potential (ERP) index of discrimination. Chinese patients with schizophrenia and Chinese healthy controls in China participated in the current study. Compared to the healthy controls, the patients with schizophrenia showed significant impairments in phonological processing skills, which in turn significantly correlated with smaller P300 effects. Thus these behavioral and electrophysiological findings in Chinese patients with schizophrenia were critically evaluated in terms of their phonological processing abilities.

Phonological recovery in Spanish developmental dyslexics through the tip-of-the-tongue paradigm.

BACKGROUND: Developmental dyslexics have difficulties accessing and retrieving the phonological form of words, in the absence of a deficit at the semantic level. The aim of this work was to study, through the Tip-of-the-tongue (TOT) paradigm, the problems of lexical access in Spanish-speaking developmental dyslexics and the relationship with their phonological awareness. METHOD: A group of developmental dyslexics (14) and other children without reading difficulties (14), aged 7 to 12, performed a picture naming task of medium and low frequency and a task of phonological awareness. RESULTS: The results indicated that dyslexic children generally show a greater number of TOT phenomena than the control group. Despite being able to provide semantic information of the drawing, they had difficulties retrieving partial phonological information. CONCLUSIONS: These results indicate that developmental dyslexic children have particular difficulty in accessing the phonological form of words, which may be interesting for the development of intervention programs for these children.

Exaggerated object affordance and absent automatic inhibition in alien hand syndrome.

Patients with alien hand syndrome (AHS) experience making apparently deliberate and purposeful movements with their hand against their will. However, the mechanisms contributing to these involuntary actions remain poorly understood. Here, we describe two experimental investigations in a patient with corticobasal syndrome (CBS) with alien hand behaviour in her right hand. First, we show that responses with the alien hand are made significantly more quickly to images of objects which afford an action with that hand compared to objects which afford an action with the unaffected hand. This finding suggests that involuntary grasping behaviours in AHS might be due to exaggerated, automatic motor activation evoked by objects which afford actions with that limb. Second, using a backwards masked priming task, we found normal automatic inhibition of primed responses in the patient's unaffected hand, but importantly there was no evidence of such suppression in the alien limb. Taken together, these findings suggest that grasping behaviours in AHS may result from exaggerated object affordance effects, which might potentially arise from disrupted inhibition of automatically evoked responses.

Developmental dyslexia: exploring how much phonological and visual attention span disorders are linked to simultaneous auditory processing deficits.

The simultaneous auditory processing skills of 17 dyslexic children and 17 skilled readers were measured using a dichotic listening task. Results showed that the dyslexic children exhibited difficulties reporting syllabic material when presented simultaneously. As a measure of simultaneous visual processing, visual attention span skills were assessed in the dyslexic children. We presented the dyslexic children with a phonological short-term memory task and a phonemic awareness task to quantify their phonological skills. Visual attention spans correlated positively with individual scores obtained on the dichotic listening task while phonological skills did not correlate with either dichotic scores or visual attention span measures. Moreover, all the dyslexic children with a dichotic listening deficit showed a simultaneous visual processing deficit, and a substantial number of dyslexic children exhibited phonological processing deficits whether or not they exhibited low dichotic listening scores. These findings suggest that processing simultaneous auditory stimuli may be impaired in dyslexic children regardless of phonological processing difficulties and be linked to similar problems in the visual modality.

Evidence for a preserved sensitivity to orthographic redundancy and an impaired access to phonological syllables in French developmental dyslexics.

To evaluate the orthographic and phonological processing skills of developmental dyslexics, we (a) examined their abilities to exploit properties of orthographic redundancy and (b) tested whether their phonological deficit extends to spelling-to-sound connections for large-grain size units such as syllables. To assess the processing skills in dyslexics, we utilized the illusory conjunction paradigm to investigate the nature of reading units in French dyslexic and control children matched in reading age. In control children, reading units were defined by both orthographic redundancy and phonological syllable information. In dyslexics, however, reading units were defined only by orthographic redundancy. Therefore, despite their impairment in reading acquisition, developmental dyslexics have the ability to encode and exploit letter frequency co-occurrences. In contrast, their access to phonological syllables from letters was impaired, suggesting that their phonological deficit extends to large grain-size phonological units.

On the importance of considering individual profiles when investigating the role of auditory sequential deficits in developmental dyslexia.

The goal of this study was to gain a better understanding of the relationship between non-verbal auditory disorders and developmental dyslexia. This question has led to conflicting results in the literature, which we argued might be due to a failure to consider the heterogeneity of dyslexic profiles. This study included three groups of adult participants: unimpaired readers and dyslexic readers with or without a phonological deficit. Auditory temporal processing deficits, as measured by stream segregation thresholds, were present in most dyslexic participants with phonological disorders. In contrast, most dyslexic participants with preserved phonological skills had normal auditory stream segregation thresholds. Overall, the present study leads to a better understanding of the relationship between phonological and sequential auditory processing disorders in developmental dyslexia. In addition, it demonstrates for the first time the importance of considering the heterogeneity of individual cognitive profiles when investigating the role of auditory deficits in developmental dyslexia.

Tongue mobility in patients with cerebral palsy.

BACKGROUND/AIM: In children with cerebral palsy speech is a big problem. Speech of these children is more or less understandable, depending on the degree of reduced mobility of articulatory organs. Reduced mobility is affected by inability to control facial grimacing and poor muscle strength when performing targeted movements. The aim of this study was to determine the mobility of tongue in patients with cerebral palsy. METHODS: The study included a sample of 34 children--patients with cerebral palsy who had been treated in the Special Hospital for the Cerebral Palsy and Developmental Neurology in Belgrade. The patients were divided according to the determined diagnosis into two groups: Quadriparesis spastica (n = 11) and Morbus Little (n = 16). The children, aged 8-12 years, had preserved intelectual abilities, and all of them had preserved hearing. The study was conducted during the period from January to September 2009. The functional state of articulatory organs in both groups was tested by the C-test that examines the anatomic structure and mobility of the articulatory organs. RESULTS: Our research showed that both groups of the patients had impaired functional state of the tongue - the most mobile articulatory organ. Also, the research showed that the functional state of the tongue was worse in children diagnosed with Quadriparesis spastica. A statistically significant correlation between the diagnosis and the functional state of the tongue, the tongue test performance and the retention of the tongue in a given position was found (r = 0.594, p < 0.005; r = 0.816, p < 0.01 and r = 0.738, p < 0.001, respectively). CONCLUSION: A large percentage of children with cerebral palsy were not able to establish control over the position of articulatory organs, especially the tongue, and its retention in a given position, all of which affect the quality of speech.

Phonological fluency is uniquely impaired in Japanese-speaking schizophrenia patients: confirmation study.

In alphabet-based language-speaking patients with schizophrenia, category fluency is disproportionately disturbed as compared with phonological fluency. Deficits in category and phonological fluency observed in Japanese patients, however, were similar. The aim of the present study was to replicate these findings by modifying the task to minimize the influence of lack of motivation and concentration in the patients. Similar deficits were found in both types of fluency in Japanese patients. Patients who speak Japanese have deficits in phonological fluency, compared with patients who speak alphabet-based languages, suggesting that the pattern of impairment in verbal fluency in schizophrenia is dependent on the specific language system.

Changes to articulatory kinematics in response to loudness cues in individuals with Parkinson's disease.

PURPOSE: Individuals with Parkinson's disease (PD) exhibit differences in displacement and velocity of the articulators as compared with older adults. The purpose of the current study was to examine effects of 3 loudness cues on articulatory movement patterns in individuals with PD. METHOD: Nine individuals diagnosed with idiopathic PD and 9 age- and sex-matched healthy controls produced sentences in 4 conditions: (a) comfortable loudness, (b) targeting 10 dB above comfortable, (c) twice as loud as comfortable, and (d) in background noise. Lip and jaw kinematics and acoustic measurements were obtained. RESULTS: Both groups significantly increased sound pressure level (SPL) in the loud conditions as compared with the comfortable condition. For the loud conditions, both groups had the highest SPL in the background noise and the 10 dB conditions, and the lowest SPL in the twice as loud condition. Control participants produced the largest opening displacement in the background noise condition and the smallest opening displacement in the twice as loud condition. Conversely, individuals with PD produced the largest opening displacement in the twice as loud condition and the smallest opening displacement in the background noise condition. CONCLUSIONS: Control participants and individuals with PD responded to cues to increase loudness in different ways. Changes in SPL may explain differences in kinematics for the control participants, but they do not explain such differences for individuals with PD.

Impairments in speech and nonspeech sound categorization in children with dyslexia are driven by temporal processing difficulties.

Auditory processing problems in persons with dyslexia are still subject to debate, and one central issue concerns the specific nature of the deficit. In particular, it is questioned whether the deficit is specific to speech and/or specific to temporal processing. To resolve this issue, a categorical perception identification task was administered in thirteen 11-year old dyslexic readers and 25 matched normal readers using 4 sound continua: (1) a speech contrast exploiting temporal cues (/bA/-/dA/), (2) a speech contrast defined by nontemporal spectral cues (/u/-/y/), (3) a nonspeech temporal contrast (spectrally rotated/bA/-/da/), and (4) a nonspeech nontemporal contrast (spectrally rotated/u/-/y/). Results indicate that children with dyslexia are less consistent in classifying speech and nonspeech sounds on the basis of rapidly changing (i.e., temporal) information whereas they are unimpaired in steady-state speech and nonspeech sounds. The deficit is thus restricted to categorizing sounds on the basis of temporal cues and is independent of the speech status of the stimuli. The finding of a temporal-specific but not speech-specific deficit in children with dyslexia is in line with findings obtained in adults using the same paradigm (Vandermosten et al., 2010, Proceedings of the National Academy of Sciences of the United States of America, 107: 10389-10394). Comparison of the child and adult data indicates that the consistency of categorization considerably improves between late childhood and adulthood, particularly for the continua with temporal cues. Dyslexic and normal readers show a similar developmental progress with the dyslexic readers lagging behind both in late childhood and in adulthood.

Pleiotropic effects of the 11p13 locus on developmental verbal dyspraxia and EEG centrotemporal sharp waves.

We recently showed genomewide linkage of centrotemporal sharp waves (CTS) in classic Rolandic epilepsy (RE) families to chromosome 11p13, and fine-mapped this locus to variants in the ELP4 gene. Speech sound disorder (SSD) is a common comorbidity in RE subjects, of unknown etiology, which co-aggregates in family members in a manner that could hypothetically be explained by shared underlying genetic risk with CTS. Furthermore, the neural mechanism of SSD is unknown, although individuals with rare, Mendelian forms of RE are described with severe verbal and oromotor apraxia. We therefore first performed genomewide linkage analysis for SSD, operationally defined as clinical history consistent with ICD-10 speech articulation disorder, in 38 families singly ascertained through a proband with RE. We tested the hypothesis of shared genetic risk with CTS at the 11p13 locus. In the second part of the study we used computerized acoustic analysis of recorded speech to test the hypothesis of dyspraxia as a mechanism for SSD in a smaller subset of RE probands and relatives. In two-point and multipoint LOD score analysis, we found that evidence for linkage to the 11p13 locus increased substantially when the phenotype was broadened from CTS to CTS/SSD. In multipoint analysis, the LOD score rose by 3.2 to HLOD 7.54 at D11S914 for CTS/SSD, the same marker at which multipoint linkage maximized for CTS alone. Non-parametric, affected-only methods in a sub-set of the data provide further confirmatory evidence for pleiotropy. In acoustic analysis there were voice-onset time abnormalities in 10/18 RE probands, 8/16 siblings and 5/15 parents, providing evidence of breakdown in the spatial/temporal properties of speech articulation consistent with a dyspraxic mechanism. The results from genetic and physiological studies suggest a pleiotropic role for the 11p13 locus in the development of both SSD and CTS, and also indicate a dyspraxic mechanism for the SSD linked to 11p13. Taken together, these data strongly support a neurodevelopmental origin for classic RE.

A treatment for dysprosody in childhood apraxia of speech.

PURPOSE: Dysprosody is considered a core feature of childhood apraxia of speech (CAS), especially impaired production of lexical stress. Few studies have tested the effects of intervention for dysprosody. This Phase II study with 3 children investigated the efficacy of a treatment targeting improved control of relative syllable durations in 3-syllable nonwords representing strong-weak (SW) and weak-strong (WS) stress patterns (e.g., BAtigu or baTIgu). Treatment sessions were structured along the principles of motor learning (PML) approach. METHOD: Three children, age 7 to 10 years, with mild to moderate CAS and normal language development participated in an intensive 3-week treatment. Within-participant designs with multiple baselines across participants and behaviors were used to examine acquisition, generalization, and maintenance of skill. RESULTS: All children improved in their ability to control relative duration of syllables in SW and WS nonwords. Improvement was also noted in control of loudness and pitch contrasts. Treatment effects generalized to untreated nonword stimuli, but minimal change was seen in production of real words. CONCLUSION: Findings support the efficacy of this approach for improving production of lexical stress contrasts. Structuring the intervention according to the PML approach likely stimulated strong maintenance and generalization effects.

Speech fluency in neurofibromatosis type 1.

Neurofibromatosis type 1 (NF1) is an autosomal dominant neurocutaneous disorder with an estimated prevalence of about 1/3000. Several authors mention the occurrence of various types of speech abnormalities associated with NF1. The present study investigated speech fluency in 21 Dutch speaking adults with NF1. Speech samples were collected in five different speaking modalities (spontaneous speech, monologue, repetition, automatic series and reading) and subsequently analysed for type, number and distribution of dysfluencies. It was found that dysfluencies are a common feature of the speech of individuals with NF1. Although stuttering appears to occur in some NF1 patients, as a group, they display a dysfluency pattern that is not identical to stuttering. Educationalobjectives: The reader will be able to (1) summarize the clinical characteristics, prevalence and genetics of neurofibromatosis type 1 (NF1) and (2) describe the dysfluent behaviour displayed by individuals with NF1 regarding frequency, type and distribution of the dysfluencies.

Relationships between speech intelligibility and word articulation scores in children with hearing loss.

PURPOSE: This investigation sought to determine whether scores from a commonly used word-based articulation test are closely associated with speech intelligibility in children with hearing loss. If the scores are closely related, articulation testing results might be used to estimate intelligibility. If not, the importance of direct assessment of intelligibility is reinforced. METHOD: Forty-four children with hearing losses produced words from the Goldman Fristoe Test of Articulation-Second Edition (Goldman & Fristoe, 2000) and sets of 10 short sentences. Correlation analyses were conducted between scores for 7 word-based predictor variables and percent-intelligible scores derived from listener judgments of stimulus sentences. RESULTS: Six of 7 predictor variables were significantly correlated with percent-intelligible scores. However, regression analysis revealed that no single predictor variable or multivariable model accounted for more than 25% of the variability in intelligibility scores. CONCLUSIONS: The findings confirm the importance of assessing connected speech intelligibility directly.

Towards a further characterization of phonological and literacy problems in Dutch-speaking children with dyslexia.

This longitudinal study examined the development of phonology and literacy in Dutch-speaking children at family risk of dyslexia and in matched controls. Measures were administered in kindergarten (before the start of formal reading instruction), in first and in third grade. Children, diagnosed with dyslexia in third grade, showed impaired phonological awareness (PA), verbal short-term memory, and rapid automatic naming ability (RAN) at all time points, with the deficit in PA aggravating over time. These children also performed more poorly in letter knowledge, word and nonword reading accuracy and speed, and spelling at each time point. Children at family risk of dyslexia who did not fulfil criteria for dyslexia, scored more poorly than low-risk controls on the literacy and phonological measures that required the most fine-grained phonological representations. This suggests that the family risk of dyslexia is continuous rather than discrete. Hierarchical regression analyses demonstrated that PA and RAN were initially the most important instigators of reading accuracy and reading speed, respectively. After 2 years of reading instruction, only RAN predicted reading speed and accuracy. Letter knowledge, reading accuracy, and reading speed also contributed to the development of PA.