Speech and Music Therapy in the Treatment of Childhood Apraxia of Speech: An Introduction and a Case Study.

PURPOSE: Speech-Music Therapy for Aphasia (SMTA), a method that combines speech therapy and music therapy, is introduced as a treatment method for childhood apraxia of speech (CAS). SMTA will be evaluated in a proof-of-principle study. The first case study is presented herein. METHOD: SMTA was evaluated in a study with a single-subject experimental design comparing 10 weeks of treatment with 2 months of no treatment. The research protocol included a pretest, baseline phase, treatment phase, posttest, no-treatment phase, and follow-up test. The participant was a boy with CAS aged 5;8 (years;months). Outcome measures were selected to reflect both intelligibility in daily communication as well as features of CAS and speech motor planning and programming. RESULTS: Results on the Intelligibility in Context Scale-Dutch (ICS-Dutch) and in the analysis of a spontaneous speech sample suggest generalization of treatment effects. Improvements were found in measures that reflect complex speech motor skills, that is, the production of consonant clusters and consistency. CONCLUSIONS: This case study showed that speech production of the participant improved after treatment with SMTA. Although intelligibility as measured with the ICS-Dutch improved over the study period, objectifying changes at the level of intelligibility in daily communication proved to be difficult. Additional measures may be necessary to gain more insight into treatment effects at this level. Overall, the results of this first case study provide sufficient support and important leads for further evaluation of SMTA in the treatment of CAS in a proof-of-principle study.

Modeling Responses to Auditory Feedback Perturbations in Adults, Children, and Children With Complex Speech Sound Disorders: Evidence for Impaired Auditory Self-Monitoring?

PURPOSE: Previous studies have found that typically developing (TD) children were able to compensate and adapt to auditory feedback perturbations to a similar or larger degree compared to young adults, while children with speech sound disorder (SSD) were found to produce predominantly following responses. However, large individual differences lie underneath the group-level results. This study investigates possible mechanisms in responses to formant shifts by modeling parameters of feedback and feedforward control of speech production based on behavioral data. METHOD: SimpleDIVA was used to model an existing dataset of compensation/adaptation behavior to auditory feedback perturbations collected from three groups of Dutch speakers: 50 young adults, twenty-three 4- to 8-year-old children with TD speech, and seven 4- to 8-year-old children with SSD. Between-groups and individual within-group differences in model outcome measures representing auditory and somatosensory feedback control gain and feedforward learning rate were assessed. RESULTS: Notable between-groups and within-group variation was found for all outcome measures. Data modeled for individual speakers yielded model fits with varying reliability. Auditory feedback control gain was negative in children with SSD and positive in both other groups. Somatosensory feedback control gain was negative for both groups of children and marginally negative for adults. Feedforward learning rate measures were highest in the children with TD speech followed by children with SSD, compared to adults. CONCLUSIONS: The SimpleDIVA model was able to account for responses to the perturbation of auditory feedback other than corrective, as negative auditory feedback control gains were associated with following responses to vowel shifts. These preliminary findings are suggestive of impaired auditory self-monitoring in children with complex SSD. Possible mechanisms underlying the nature of following responses are discussed.

Music and musical elements in the treatment of childhood speech sound disorders: A systematic review of the literature.

PURPOSE: Music-based interventions are used in the treatment of childhood speech sound disorders (SSDs). Hypotheses on working mechanisms are being developed, focussing on shared neural processes. However, evidence of the effect of treatment with musical elements in SSDs in children is lacking. This study reviews the literature regarding the use of music-based interventions in the treatment of childhood SSDs. METHOD: A systematic search in six databases was conducted, yielding 199 articles, eight of which met the inclusion criteria. Included articles were reviewed on study characteristics, patient characteristics, interventions, outcomes and methodological quality. RESULT: This review included four case studies, three single-subject design studies and one cohort study. Seven studies reported positive outcomes on speech production, but outcome measures in the four studies with experimental design were not all aimed at the level of speech (motor) processes. Methodological quality was sufficient in one study. CONCLUSION: Seven out of eight studies in this review report positive outcomes of music-based interventions in the treatment of SSDs. However, these outcomes are not sufficiently supported by evidence due to insufficient methodological quality. Suggestions for improving methodological quality in future research are presented.

Process-Oriented Profiling of Speech Sound Disorders.

The differentiation between subtypes of speech sound disorder (SSD) and the involvement of possible underlying deficits is part of ongoing research and debate. The present study adopted a data-driven approach and aimed to identify and describe deficits and subgroups within a sample of 150 four to seven-year-old Dutch children with SSD. Data collection comprised a broad test battery including the Computer Articulation Instrument (CAI). Its tasks Picture Naming (PN), NonWord Imitation (NWI), Word and NonWord Repetition (WR; NWR) and Maximum Repetition Rate (MRR) each render a variety of parameters (e.g., percentage of consonants correct) that together provide a profile of strengths and weaknesses of different processes involved in speech production. Principal Component Analysis on the CAI parameters revealed three speech domains: (1) all PN parameters plus three parameters of NWI; (2) the remaining parameters of NWI plus WR and NWR; (3) MRR. A subsequent cluster analysis revealed three subgroups, which differed significantly on intelligibility, receptive vocabulary, and auditory discrimination but not on age, gender and SLPs diagnosis. The clusters could be typified as three specific profiles: (1) phonological deficit; (2) phonological deficit with motoric deficit; (3) severe phonological and motoric deficit. These results indicate that there are different profiles of SSD, which cover a spectrum of degrees of involvement of different underlying problems.

Maximum repetition rate in a large cross-sectional sample of typically developing Dutch-speaking children.

Purpose: The current study aims to provide normative data for the maximum repetition rate (MRR) development of Dutch-speaking children based on a large cross-sectional study using a standardised protocol.Method: A group of 1014 typically developing children aged 3;0 to 6;11 years performed the MRR task of the Computer Articulation Instrument (CAI). The number of syllables per second was calculated for mono-, bi-, and trisyllabic sequences (MRR-pa, MRR-ta, MRR-ka, MRR-pata, MRR-taka, MRR-pataka). A two-way mixed ANOVA was conducted to compare the effects of age and gender on MRR scores in different MRR sequences.Result: The data analysis showed that overall MRR scores were affected by age group, gender and MRR sequence. For all MRR sequences the MRR increased significantly with age. MRR-pa was the fastest sequence, followed by respectively MRR-ta, MRR-pata, MRR-taka, MRR-ka and MRR-pataka. Overall MRR scores were higher for boys than for girls, for all MRR sequences.Conclusion: This study presents normative data of MRR of Dutch-speaking children aged 3;0 to 6;11 years. These norms might be useful in clinical practice to differentiate children with speech sound disorders from typically developing children. More research on this topic is necessary. It is also suggested to collect normative data for other individual languages, using the same protocol.

Differential Diagnosis of Childhood Apraxia of Speech Compared to Other Speech Sound Disorders: A Systematic Review.

Purpose The aim of this study was to determine the discriminative features that might contribute to differentiation of childhood apraxia of speech (CAS) from other speech sound disorders (SSDs). Method A comprehensive literature search was conducted for articles or doctoral dissertations that included ≥ 1 child with CAS and ≥ 1 child with SSD. Of 2,071 publications screened, 53 met the criteria. Articles were assessed for (a) study design and risk of bias; (b) participant characteristics and confidence in diagnosis; and (c) discriminative perceptual, acoustic, or kinematic measures. A criterion was used to identify promising studies: American Academy of Neurology study design (Class III+), replicable participant descriptions and adequate confidence in diagnosis (≥ 3), and ≥ 1 discriminative and reliable measure. Results Over 75% of studies were retrospective, case-control designs and/or assessed English-speaking children. Many studies did not fully describe study design and quality. No studies met the Class I (highest) quality rating according to American Academy of Neurology guidelines. CAS was mostly compared to speech delay/phonological disorder. Only six studies had diagnostic confidence ratings of 1 (best). Twenty-six studies reported discriminative perceptual measures, 14 reported discriminative acoustic markers, and four reported discriminative kinematic markers. Measures were diverse, and only two studies directly replicated previous findings. Overall, seven studies met the quality criteria, and another eight nearly met the study criteria to warrant further investigation. Conclusions There are no studies of the highest diagnostic quality. There are 15 studies that can contribute to further diagnostic efforts discriminating CAS from other SSDs. Future research should utilize careful diagnostic design, support replication, and adhere to standard reporting guidelines. Supplemental Material https://doi.org/10.23641/asha.13158149.

Clinical Reasoning for Speech Sound Disorders: Diagnosis and Intervention in Speech-Language Pathologists' Daily Practice.

Purpose This study aims to give an insight in clinical reasoning (diagnosis and intervention) of speech-language pathologists (SLPs) in the Netherlands for children with speech sound disorder (SSD). Method The study featured a mixed-method (qualitative and quantitative) design. Semistructured interviews containing nondirective, open-ended questions were conducted with 33 SLPs, which were analyzed using a constant comparative analysis. Other SLPs (137) filled out a questionnaire on the same topics. Multiple-choice questions were analyzed by descriptive frequencies, while open-ended questions were analyzed thematically. Results The results indicate that SLPs use a variety of assessments to diagnose SSD, complemented by observation and, often, case history. In total, 85 different diagnostic labels were reported. The choice of intervention is based on what is appealing to the child and what matches his or her age as well as on the specific diagnosis and severity. Interventions are used for multiple speech disorders, and according to SLPs, parents play a large role in diagnostics and intervention. Conclusion These results reveal the need for (a) a clear and consistent terminology of diagnoses in the field of pediatric SSD, (b) a fast and easy-to-administer comprehensive differential diagnostic instrument in combination with an instrument to assess participation in everyday life, and (c) a tool to conduct a case history online.

Compensatory and adaptive responses to real-time formant shifts in adults and children.

Auditory feedback plays an important role in speech motor learning, yet, little is known about the strength of motor learning and feedback control in speech development. This study investigated compensatory and adaptive responses to auditory feedback perturbation in children (aged 4-9 years old) and young adults (aged 18-29 years old). Auditory feedback was perturbed by near-real-time shifting F1 and F2 of the vowel /ɪː/ during the production of consonant-vowel-consonant words. Children were able to compensate and adapt in a similar or larger degree compared to young adults. Higher token-to-token variability was found in children compared to adults but not disproportionately higher during the perturbation phases compared to the unperturbed baseline. The added challenge to auditory-motor integration did not influence production variability in children, and compensation and adaptation effects were found to be strong and sustainable. Significant group differences were absent in the proportions of speakers displaying a compensatory or adaptive response, an amplifying response, or no consistent response. Within these categories, children produced significantly stronger compensatory, adaptive, or amplifying responses, which could be explained by less-ingrained existing representations. The results are interpreted as both auditory-motor integration and learning capacities are stronger in young children compared to adults.

Testing hypotheses about the underlying deficit of apraxia of speech through computational neural modelling with the DIVA model.

Purpose: A recent behavioural experiment featuring a noise masking paradigm suggests that Apraxia of Speech (AOS) reflects a disruption of feedforward control, whereas feedback control is spared and plays a more prominent role in achieving and maintaining segmental contrasts. The present study set out to validate the interpretation of AOS as a possible feedforward impairment using computational neural modelling with the DIVA (Directions Into Velocities of Articulators) model.Method: In a series of computational simulations with the DIVA model featuring a noise-masking paradigm mimicking the behavioural experiment, we investigated the effect of a feedforward, feedback, feedforward + feedback, and an upper motor neuron dysarthria impairment on average vowel spacing and dispersion in the production of six/bVt/speech targets.Result: The simulation results indicate that the output of the model with the simulated feedforward deficit resembled the group findings for the human speakers with AOS best.Conclusion: These results provide support to the interpretation of the human observations, corroborating the notion that AOS can be conceptualised as a deficit in feedforward control.

Assessment of Childhood Apraxia of Speech: A Review/Tutorial of Objective Measurement Techniques.

Background With respect to the clinical criteria for diagnosing childhood apraxia of speech (commonly defined as a disorder of speech motor planning and/or programming), research has made important progress in recent years. Three segmental and suprasegmental speech characteristics-error inconsistency, lengthened and disrupted coarticulation, and inappropriate prosody-have gained wide acceptance in the literature for purposes of participant selection. However, little research has sought to empirically test the diagnostic validity of these features. One major obstacle to such empirical study is the fact that none of these features is stated in operationalized terms. Purpose This tutorial provides a structured overview of perceptual, acoustic, and articulatory measurement procedures that have been used or could be used to operationalize and assess these 3 core characteristics. Methodological details are reviewed for each procedure, along with a short overview of research results reported in the literature. Conclusion The 3 types of measurement procedures should be seen as complementary. Some characteristics are better suited to be described at the perceptual level (especially phonemic errors and prosody), others at the acoustic level (especially phonetic distortions, coarticulation, and prosody), and still others at the kinematic level (especially coarticulation, stability, and gestural coordination). The type of data collected determines, to a large extent, the interpretation that can be given regarding the underlying deficit. Comprehensive studies are needed that include more than 1 diagnostic feature and more than 1 type of measurement procedure.

Profiling Speech Sound Disorders for Clinical Validation of the Computer Articulation Instrument.

Purpose The current article presents data from 2 studies on clinical groups of children referred for speech assessment. The aims of these studies are to validate the Computer Articulation Instrument (CAI) with the known-group validation method and to determine the differential diagnostic power of the resulting speech profiles. Method Study 1 examined known-group validity by comparing the scores of 93 children diagnosed with speech-language difficulties on the picture naming (PN) task of the CAI with intelligibility judgments given by speech-language pathologists. In Study 2, the speech profiles of 41 children diagnosed with speech sound disorders (SSDs), consisting of 4-6 factor scores extracted from the 4 tasks of the CAI, namely, PN, nonword imitation (NWI), word and nonword repetition, and maximum repetition rate (MRR), were validated against clinical judgments of severity of the SSD given by speech-language pathologists. Results In Study 1, a repeated-measures analysis of variance revealed a significant effect of intelligibility level on the PN performance of the CAI and there were highly significant correlations between intelligibility and PN performance in the expected direction. Neither intelligibility level nor PN performance was related to nonverbal intelligence and language scores. The analysis of variance and a series of t tests in Study 2 revealed significant differences between the moderate and severe groups for the CAI factors based on PN and NWI and the bisyllabic and trisyllabic sequences of MRR, but not for the factor word and nonword proportion of whole-word variability based on word and nonword repetition, and the monosyllabic sequences of MRR. These results suggest that, especially, the tasks PN, NWI, and the bisyllabic and trisyllabic sequences of MRR are most sensitive for diagnosing SSDs. Conclusions The findings of these 2 studies support the known-group validity of the CAI. Together with the results of a previous study of our group on reliability and validity ( van Haaften et al., 2019 ), we can conclude that the CAI is a reliable and valid tool for assessment of children with SSDs.

A Psycholinguistic Framework for Diagnosis and Treatment Planning of Developmental Speech Disorders.

BACKGROUND: Differential diagnosis and treatment planning of developmental speech disorders (DSD) remains a major challenge in paediatric speech-language pathology. Different classification systems exist, in which subtypes are differentiated based on their theoretical cause and in which the definitions generally refer to speech production processes. Accordingly, various intervention methods have been developed aiming at different parts of the speech production process. Diagnostic classification in these systems, however, is primarily based on a description of behavioural speech symptoms rather than on underlying deficits. PURPOSE: In this paper, we present a process-oriented approach to diagnosis and treatment planning of DSD. Our framework comprises two general diagnostic categories: developmental delay and developmental disorder. Within these categories, treatment goals/targets and treatment methods are formulated at the level of processes and rules/representations. CONCLUSION: A process-oriented approach to diagnosis and treatment planning holds important advantages, offering direct leads for treatment aimed at the underlying impairment, tailored to the specific needs of the individual and adjusted to the developmental trajectory.

A Standardized Protocol for Maximum Repetition Rate Assessment in Children.

BACKGROUND/AIMS: Maximum repetition rate (MRR) is often used in the assessment of speech motor performance in older children and adults. The present study aimed to evaluate a standardized protocol for MRR assessment in young children in Dutch. METHODS: The sample included 1,524 children of 2-7 years old with no hearing difficulties and Dutch spoken in their nursery or primary school and was representative for children in the Netherlands. The MRR protocol featured mono-, tri-, and bisyllabic sequences and was computer-implemented to maximize standardization. RESULTS: Less than 50% of the 2-year-olds could produce >1 monosyllabic sequence correctly. Children who could not correctly produce ≥2 monosyllabic sequences could not produce any of the multisyllabic sequences. The effect of instruction ("faster" and "as fast as possible") was small, and multiple attempts yielded a faster MRR in only 20% of the cases. MRRs did not show clinically relevant differences when calculated over different numbers of repeated syllables. CONCLUSIONS: The MRR protocol is suitable for children of 3 years and older. If children cannot produce at least 2 of the monosyllabic sequences, the multisyllabic tasks should be omitted. Furthermore, all fast attempts of each sequence should be analyzed to determine the fastest MRR.

Speech Impairment in Boys With Fetal Alcohol Spectrum Disorders.

Background: Fetal alcohol spectrum disorders (FASD) are a highly prevalent spectrum of patterns of congenital defects resulting from prenatal exposure to alcohol. Approximately 90% of the cases involve speech impairment. Yet, to date, no detailed symptom profiles nor dedicated treatment plans are available for this population. Purpose: This study set out to chart the speech and speech motor characteristics in boys with FASD to profile the concomitant speech impairment and identify possible underlying mechanisms. Method: Ten boys with FASD (4.5-10.3 years old) and 26 typically developing children (4.1-8.7 years old; 14 boys, 12 girls) participated in the study. Speech production and perception, and oral motor data were collected by standardized tests. Results: The boys with FASD showed reduced scores on all tasks as well as a deviant pattern of correlations between production and perception tasks and intelligibility compared with the typically developing children. Speech motor profiles showed specific problems with nonword repetition and tongue control. Conclusions: Findings indicate that the speech impairment in boys with FASD results from a combination of deficits in multiple subsystems and should be approached as a disorder rather than a developmental delay. The results suggest that reduced speech motor planning/programming, auditory discrimination, and oral motor abilities should be considered in long-term, individually tailored treatment.

Impact of communication partner familiarity and speech accuracy on parents' ratings of their child for the Intelligibility in Context Scale: Dutch.

PURPOSE: The aim of this study was to examine the role of familiarity of a communication partner on Dutch parents' perception of their child's intelligibility, for children with typically-developing speech (TD) or speech sound disorder (SSD). METHOD: Participants were 67 Dutch-speaking children (48-84 months), 48 with TD and 19 with SSD. Item scores on the parent-rated Intelligibility in Context Scale: Dutch (ICS-NL) were compared between groups and related to naive listeners' ratings of children's intelligibility (IR), and a measure of speech accuracy (i.e. percentage of consonants correct-adjusted, PCC-A). RESULT: Statistical analysis yielded a significant Group by Familiarity interaction on the ICS-NL. Parents rated the intelligibility of their child with SSD as higher with more familiar communication partners than less familiar, more so than parents of children with TD. In the SSD group, IR was more strongly correlated with ICS-NL item scores for less familiar partners. PCC-A was only correlated with ICS-NL item 7 (strangers). CONCLUSION: Parents perceive their children as more intelligible with people in close relationships, likely due to their higher familiarity with the child's speech. Children's relationships should be considered with respect to communicative participation. PCC-A may be a less reliable predictor of participation in family and community life.

Effectiveness of speech therapy in adults with intellectual disabilities.

BACKGROUND: This study investigated the effect of speech therapy in a heterogeneous group of adults with intellectual disability. METHOD: Thirty-six adults with mild and moderate intellectual disabilities (IQs 40-70; age 18-40 years) with reported poor speech intelligibility received tailored training in articulation and listening skills delivered in two 3-month periods. Pre- to post-changes in speech intelligibility and receptive vocabulary were assessed using standardized tasks. RESULTS: The results showed a positive effect of treatment on speech intelligibility and receptive vocabulary, irrespective of severity of intellectual disability, hearing loss and intellectual disability aetiology. CONCLUSIONS: Speech therapy for people with intellectual disability can be effective at adult age and hearing loss should not prevent treatment. Continued attention to speech can help augment verbal communication skills in this population.

Speech Characteristics and Intelligibility in Adults with Mild and Moderate Intellectual Disabilities.

PURPOSE: Adults with intellectual disabilities (ID) often show reduced speech intelligibility, which affects their social interaction skills. This study aims to establish the main predictors of this reduced intelligibility in order to ultimately optimise management. METHOD: Spontaneous speech and picture naming tasks were recorded in 36 adults with mild or moderate ID. Twenty-five naïve listeners rated the intelligibility of the spontaneous speech samples. Performance on the picture-naming task was analysed by means of a phonological error analysis based on expert transcriptions. RESULTS: The transcription analyses showed that the phonemic and syllabic inventories of the speakers were complete. However, multiple errors at the phonemic and syllabic level were found. The frequencies of specific types of errors were related to intelligibility and quality ratings. CONCLUSIONS: The development of the phonemic and syllabic repertoire appears to be completed in adults with mild-to-moderate ID. The charted speech difficulties can be interpreted to indicate speech motor control and planning difficulties. These findings may aid the development of diagnostic tests and speech therapies aimed at improving speech intelligibility in this specific group.

Cognitive Functions in Childhood Apraxia of Speech.

PURPOSE: Childhood apraxia of speech (CAS) is diagnosed on the basis of specific speech characteristics, in the absence of problems in hearing, intelligence, and language comprehension. This does not preclude the possibility that children with this speech disorder might demonstrate additional problems. METHOD: Cognitive functions were investigated in 3 domains: complex sensorimotor and sequential memory functions, simple sensorimotor functions, and nonrelated control functions. Seventeen children with CAS were compared with 17 children with normal speech development at 2 occasions within 15 months. RESULTS: The children with CAS showed overall lower scores but similar improvement at Occasion 2 compared with the typically developing controls, indicating an overall delay in the development of cognitive functions. However, a specific deviant development in sequential abilities was found as well, indicated by significantly lower scores at Occasion 2 as compared with younger control children at Occasion 1. Furthermore, the scores on the complex sensorimotor and sequential memory tasks were significantly correlated with the severity of the speech impairment. CONCLUSIONS: These results suggest that CAS involves a symptom complex that not only comprises errors of sequencing speech movements but implicates comorbidity in nonverbal sequential functioning in most children with CAS.

Auditory feedback perturbation in children with developmental speech sound disorders.

BACKGROUND/PURPOSE: Several studies indicate a close relation between auditory and speech motor functions in children with speech sound disorders (SSD). The aim of this study was to investigate the ability to compensate and adapt for perturbed auditory feedback in children with SSD compared to age-matched normally developing children. METHOD: 17 normally developing children aged 4.1-8.7 years (mean=5.5, SD=1.4), and 11 children with SSD aged 3.9-7.5 years (mean=5.1, SD=1.0) participated in the study. Auditory feedback was perturbed by real-time shifting the first and second formant of the vowel /e/ during the production of CVC words in a five-step paradigm (practice/familiarization; start/baseline; ramp; hold; end/release). RESULTS: At the group level, the normally developing children were better able to compensate and adapt, adjusting their formant frequencies in the direction opposite to the perturbation, while the group of children with SSD followed (amplifying) the perturbation. However, large individual differences lie underneath. Furthermore, strong correlations were found between the amount of compensation and performance on oral motor movement non-word repetition tasks. CONCLUSIONS: Results suggested that while most children with SSD can detect incongruencies in auditory feedback and can adapt their target representations, they are unable to compensate for perturbed auditory feedback. These findings suggest that impaired auditory-motor integration may play a key role in SSD. LEARNING OUTCOMES: The reader will be able to: (1) describe the potential role of auditory feedback control in developmental speech disorders (SSD); (2) identify the neural control subsystems involved in feedback based speech motor control; (3) describe the differences between compensation and adaptation for perturbed auditory feedback; (4) explain why auditory-motor integration may play a key role in SSD.