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.

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.

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.

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.

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.

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.

Foreign accent syndrome as a developmental motor speech disorder.

INTRODUCTION: Foreign Accent Syndrome (FAS) is a relatively rare motor speech disorder in which the pronunciation of a patient is perceived by listeners of the same language community as distinctly foreign. FAS has been well documented in adult patients with etiologically heterogeneous, though mostly vascular brain lesions affecting the motor speech network of the language dominant hemisphere. In addition, reports exist of adult patients in whom FAS was due to a psychiatric illness. Although FAS has been reported in children, such accounts are rare and have remained largely anecdotal in that there have been no formally documented cases of FAS as a developmental motor speech disorder. METHODS AND RESULTS: For the first time, we describe the clinical, cognitive and neurolinguistic findings in two patients who in the absence of a history of psychiatric illness or acquired brain damage already presented with FAS at an early stage of speech and language development. In the first patient "developmental FAS" was associated with a dysharmonic distribution of neurocognitive test results indicating slight underdevelopment of visuo-spatial skills and visual memory. The second patient presented with "developmental FAS" associated with specific language impairment (SLI). Independent support for a diagnosis of FAS in both patients was obtained in an accent attribution experiment in which groups of native speakers of (Belgian) Dutch assessed the type of foreign accent of a sample of the patients' conversational speech. Both patients were judged as non-native speakers of Dutch by the majority of participants who predominantly identified the accent as French. CONCLUSION: This paper for the first time documents two patients who presented with FAS on a developmental basis. The finding that FAS does not only occur in the context of acquired brain damage or psychogenic illness but also exists as developmental motor speech impairment requires a re-definition of FAS as a clinical syndrome.

Articulatory agility in cognitive decline.

OBJECTIVE: Motor speech disorders are believed to be uncommon in early Alzheimer's disease (AD). However, data from maximum performance tests of motor speech function in AD and related disorders are virtually nonexistent. The aim of this study was to make such data available. MATERIALS AND METHODS: Sequential speech motion rate was analyzed in 236 memory clinic patients with different levels of cognitive functioning. RESULTS: Sequential speech motion rate was moderately but significantly decreased in mild dementia in AD. About 10% of AD and mild cognitive impairment cases had markedly decreased rates. Rates were strongly reduced in progressive nonfluent aphasia, whereas semantic dementia did not differ from subjective cognitive impairment. Frontotemporal dementia had lower rates than AD. CONCLUSIONS: A proportion of patients with cognitive decline has markedly reduced articulatory agility. The cause of this reduction in some patients with mild cognitive impairment and mild AD is unknown. Semantic dementia is not associated with impaired articulatory agility.

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.

Auditory processing disorder in children with reading disabilities: effect of audiovisual training.

Reading disability is associated with phonological problems which might originate in auditory processing disorders. The aim of the present study was 2-fold: first, the perceptual skills of average-reading children and children with dyslexia were compared in a categorical perception task assessing the processing of a phonemic contrast based on voice onset time (VOT). The medial olivocochlear (MOC) system, an inhibitory pathway functioning under central control, was also explored. Secondly, we investigated whether audiovisual training focusing on voicing contrast could modify VOT sensitivity and, in parallel, induce MOC system plasticity. The results showed an altered voicing sensitivity in some children with dyslexia, and that the most severely impaired children presented the most severe reading difficulties. These deficits in VOT perception were sometimes accompanied by MOC function abnormalities, in particular a reduction in or even absence of the asymmetry in favour of the right ear found in average-reading children. Audiovisual training significantly improved reading and shifted the categorical perception curve of certain children with dyslexia towards the average-reading children's pattern of voicing sensitivity. Likewise, in certain children MOC functioning showed increased asymmetry in favour of the right ear following audiovisual training. The training-related improvements in reading score were greatest in children presenting the greatest changes in MOC lateralization. Taken together, these results confirm the notion that some auditory system processing mechanisms are impaired in children with dyslexia and that audiovisual training can diminish these deficits.

Square-wave jerks and smooth pursuit impairment as subtle early signs of brain involvement in Langerhans' cell histiocytosis.

Central nervous system (CNS) involvement in Langerhans' cell histiocytosis (LCH) has been described as a progressive neurological disorder marked by motor and cognitive decline. Detailed analysis of ocular motor abnormalities is lacking. We report on a 60-year-old male with histologically confirmed LCH who developed oscillopsia and gait ataxia over a 1-year period. Eye movements recorded with infrared oculography revealed a high rate of square-wave jerks (SWJ) with frequencies of 41 min(-1) on average and amplitudes between 1 degrees and 7 degrees , as well as marked impairment of smooth tracking of sinusoidally moving targets. Furthermore, static posturography disclosed increased body sway, with an abnormally high sway path. The initial brain MRI was unremarkable. Due to the presumed cerebellar dysfunction we performed a second MRI 1 year later that disclosed deep cerebellar lesions compatible with LCH relapse within the CNS. The abnormal high SWJ rate and the impaired smooth pursuit performance correctly heralded later involvement of the cerebellum anticipating lesion appearance in the MRI.

Somatosensory-evoked magnetic fields in examining lip area in speech-disordered children.

Magnetoencephalography (MEG) is a modern neurophysiological method to study brain activation after sensory stimulation. We aimed at determining the feasibility of MEG and somatosensory-evoked magnetic fields (SEFs) in assessing the lip area in speech-disordered children undergoing oral plate therapy (OPTH) to improve their articulation. Seven subjects (age range, 6-11 years) participated in the study. The speech was perceptually assessed, and the SEFs to tactile stimulation of the lip area were recorded before and after OPTH. Two patients did not attend the posttreatment MEG recording. Clinical perceptual analysis showed remarkable improvement of speech of the studied children after OPTH. Somatosensory-evoked magnetic fields were successfully recorded in 4 of these children, but no constant changes in the responses were found after the therapy.With this small number of patients, the possible modifications in the functioning of the cortical somatosensory area of the lip after OPTH remained undetected. The present method is, however, technically applicable in studying cortical responses to lip stimulation in speech-disordered children. Further studies using stimulation inside the mouth may provide more insight to the cortical effects of OPTH.

[Study of causal factors of reading impairment in a sample of 1062 7 to 8-year-old children]. / Etude des facteurs liés aux difficultés d'apprentissage de la lecture. A partir d'un échantillon de 1062 enfants de seconde année d'école élémentaire.

OBJECTIVE: From an original large sample of 1062 7 to 8-year-old children, reading skills were assessed and found to be highly linked with socioeconomic status (SES). The purpose of the present study was to further determine underlying medical, sociocultural, cognitive and behavioural factors explaining the diversity of reading skills and the influence of SES. METHOD: Individual testing among low-SES children identified 100 poor readers, 50 typical readers and 31 children with intermediate reading scores. All 3 groups underwent a thorough assessment, including a medical evaluation, a full cognitive battery, a structured parental interview and behavioural questionnaire. Logistic regression was used to demonstrate the variables predicting reading score outcome. RESULTS: None of the medical factors studied was statistically related to reading scores. Due to the methodology buffering the impact of SES sociocultural variables, such as parental levels of education, parental occupation, as well as familial income were weak, but statistically significant predictors. The strongest variables were phonological abilities and symptoms of attention disorders. In a final regression model, phonological awareness, level of mother's education and attention explained the differences in reading skills. CONCLUSIONS: These results, which are unique in France, are similar to existing data in the literature. They support the need to conceptualize an early screening programme to detect reading difficulties and to promote an intervention based on phonological processing and decoding in low-SES environments.

Exploring the dynamics of aphasic word production using the picture-word interference task: a case study.

In this study, we use an auditory picture-word interference task to examine an anomic individual, NP. NP produced semantic errors in picture naming, but his comprehension was relatively well preserved. In the task, pictures to be named were accompanied by semantically, phonologically or unrelated distractors, presented at onsets ranging from -200ms (before target) to +400ms (after target). Naming latencies were measured. A group of 12 older controls showed semantic interference (slower latencies with semantic than with unrelated distractors), which was significant at -200ms, and steadily diminished across later onsets. In contrast, at 0ms, NP showed powerful semantic facilitation. There were no significant semantic effects at other onsets, but the trends, particularly at later onsets, were towards interference. Phonological effects for NP were in the same direction as for controls (facilitation) but were of greater magnitude. Indeed, NP showed a reliable facilitatory effect at 0ms (and trends at -200ms and +200ms), but a similar trend in controls failed to reach significance. Within recent models of this task, in which semantic facilitation effects are attributed to an early, pre-lexical semantic processing stage, NP's pattern indicates that semantic processing is abnormally prolonged. The phonological facilitation effects are also consistent with this interpretation. We discuss their implications and future applications of the task to aphasia.

Non-word repetition in children with specific language impairment: a deficit in phonological working memory or in long-term verbal knowledge?

In this study we investigated the effects of long-term memory (LTM) verbal knowledge on short-term memory (STM) verbal recall in a sample of Italian children affected by different subtypes of specific language impairment (SLI). The aim of the study was to evaluate if phonological working memory (PWM) abilities of SLI children can be supported by LTM linguistic representations and if PWM performances can be differently affected in the various subtypes of SLI. We tested a sample of 54 children affected by Mixed Receptive-Expressive (RE), Expressive (Ex) and Phonological (Ph) SLI (DSM-IV - American Psychiatric Association, 1994) by means of a repetition task of words (W) and non-words (NW) differing in morphemic structure [morphological non-words (MNW), consisting of combinations of roots and affixes - and simple non-words - with no morphological constituency]. We evaluated the effects of lexical and morpho-lexical LTM representations on STM recall by comparing the repetition accuracy across the three types of stimuli. Results indicated that although SLI children, as a group, showed lower repetition scores than controls, their performance was affected similarly to controls by the type of stimulus and the experimental manipulation of the non-words (better repetition of W than MNW and NW, and of MNW than NW), confirming the recourse to LTM verbal representations to support STM recall. The influence of LTM verbal knowledge on STM recall in SLI improved with age and did not differ among the three types of SLI. However, the three types of SLI differed in the accuracy of their repetition performances (PMW abilities), with the Phonological group showing the best scores. The implications for SLI theory and practice are discussed.

Speech development patterns and phonological awareness in preschool children.

To examine the association between speech production and early literacy skills, this study of 102 preschool children looked at phonological awareness in relation to whether children were delayed, typical, or advanced in their articulation of consonants. Using a developmental typology inspired by some of the literature on speech development (Kahn and Lewis, The Kahn-Lewis phonological analysis, 1986; Shriberg, Journal of Speech and Hearing Research 36(1):105-140, 1993a), we found that failure to master the early-8 consonants and a greater prevalence of certain types of production errors were associated with deficient phonological awareness. We also found that children who made no consonant errors had advanced phonological awareness relative to other children in the sample. In all cases, both productive speech patterns and speech errors were more closely linked with rhyme awareness than with phoneme awareness. The association between speech production and rhyme awareness may provide some new directions for the early preschool assessment of risk for reading problems.

Speech perception in preschoolers at family risk for dyslexia: relations with low-level auditory processing and phonological ability.

We tested categorical perception and speech-in-noise perception in a group of five-year-old preschool children genetically at risk for dyslexia, compared to a group of well-matched control children and a group of adults. Both groups of children differed significantly from the adults on all speech measures. Comparing both child groups, the risk group presented a slight but significant deficit in speech-in-noise perception, particularly in the most difficult listening condition. For categorical perception a marginally significant deficit was observed on the discrimination task but not on the identification task. Speech parameters were significantly related to phonological awareness and low-level auditory measures. Results are discussed within the framework of a causal model where low-level auditory problems are hypothesized to result in subtle speech perception problems that might interfere with the development of phonology and reading and spelling ability.

Overall intelligibility, language, articulation, voice and resonance characteristics in a child with Shprintzen-Goldberg syndrome.

OBJECTIVE: Recognition of the phenotypic spectrum and prognosis of a genetic disorder is critical to proper patient care. A 7-year-old boy with Sphrintzen-Goldberg syndrome (SGS) was studied to investigate speech, language and voice patterns associated with this syndrome. METHODS: The child's language (expressive and receptive) and speech was characterized with regard to overall intelligibility, articulation (phonetic and phonological errors), voice (flexible videolaryngostroboscopy, quality, pitch and loudness) and resonance (type of disorders). RESULTS: Based on this detailed study the most striking communication characteristics in this child with SGS appear to be a delayed speech and language onset, an expressive and receptive language disorder, a moderately impaired speech intelligbility, relatively good phonetic but poorer phonological abilities, an oral hypotonia, a high-pitched soft voice and a slight hypernasality. CONCLUSIONS: The explanation for this communication disorder is not completely straightforward. It is not clear either to what extent the present case can be considered as typical for SGS. Only more data will allow to determine whether or not SGS is associated with a typical syndrome specific pattern of communication disorders. Not only detailed speech and language analyses of additional cases of SGS are necessary, but also studies that compare the speech and language of individuals with SGS with that of individuals with other genetic syndrome.

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.

Developmental dyslexia: specific phonological deficit or general sensorimotor dysfunction?

Dyslexia research now faces an intriguing paradox. It is becoming increasingly clear that a significant proportion of dyslexics present sensory and/or motor deficits; however, as this 'sensorimotor syndrome' is studied in greater detail, it is also becoming increasingly clear that sensory and motor deficits will ultimately play only a limited role in a causal explanation of specific reading disability.