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Background: Accurate nonword repetition (NWR) is contingent on many underlying skills, including encoding, memory and motor planning and programming. Though vowel errors are frequently associated with childhood apraxia of speech (CAS), several recent studies have found that children with developmental language disorder (DLD) produce high rates of vowel errors in NWR tasks. Aims: This retrospective analysis explored whether the overall frequency and types of vowel errors produced in NWR distinguish children with DLD, children with CAS, children with speech sound disorder (SSD) and children with typical development (TD). Methods and Procedure: We present data for 24 children (six per DLD, CAS, SSD and TD groups), ranging in age from 50–92 months. Children with DLD, CAS and SSD showed similar articulation scores and children with DLD and children with CAS showed similar expressive language scores. Total vowel errors, total monophthong errors, monophthong substitutions, diphthongization errors, total diphthong errors, diphthong substitutions and diphthong reduction errors were calculated by syllable length and group. Repeated measures analyses of variance were used to examine group differences. Outcomes and Results: Children with DLD and children with CAS produced a higher frequency of total vowel errors compared to children with TD. Children with DLD produced more total monophthong errors than children with TD. Children with DLD and children with CAS produced more total diphthong errors than children with TD. For children with DLD, these were characterised by diphthong substitutions. For children with CAS, these were characterised by diphthong substitutions and diphthong reduction errors. For all measures, error rates in children with SSD did not significantly differ from any of the other three groups. Conclusion: Preliminary evidence indicates that children with DLD and children with CAS both show high rates of vowel errors in NWR tasks and weaknesses in encoding and memory. For children with CAS, additional motor planning difficulties are associated with an increased likelihood to reduce diphthongs. Children with SSD show more mild processing difficulties than children with DLD and children with CAS, though they do not perform as well as TD peers. Future work should replicate and further specify the processing weaknesses that affect vowel accuracy in NWR tasks in a larger sample. WHAT THIS PAPER ADDS: What is already known on the subject: Nonword repetition (NWR) tasks are often included in diagnostic batteries to identify children with developmental language disorder (DLD). Poor performance on these tasks have historically been attributed to phonological working memory deficits in children with DLD. However, repeating nonwords relies on a number of underlying processing skills and many of these skills are affected to varying degrees in children with speech and language disorders. An in‐depth analysis of vowel errors has the potential to reveal the shared as well as specific underlying processing weaknesses in children with DLD, children with childhood apraxia of speech (CAS) and children with speech sound disorder (SSD). What this paper adds to existing knowledge: We found that children with DLD and children with CAS show low vowel competence compared to children with typical development. A nuanced examination of vowel error types further revealed that children with DLD and children with CAS show weaknesses in encoding and memory. Motor planning and programming weaknesses were unique to CAS. Children with SSD show more mild processing deficits and their performance did not significantly differ from any of the other three groups. What are the clinical implications of this work?: Examining the types of vowel errors produced by children with DLD, children with CAS and children with SSD in NWR allows us to further specify the underlying processing weaknesses that differentiate these three groups. This research informs theoretical accounts of language processing in children with different types of speech and language disorders and has the potential to improve the diagnostic utility of NWR tasks. [ABSTRACT FROM AUTHOR]

Background: Phonological difficulties are prevalent in children with speech and/or language disorders and may hamper their later language outcomes and academic achievements. These children often form a significant proportion of speech and language therapists' caseloads. There is a shortage of information on evidence‐based interventions for improving phonological skills in children and adolescents with speech and language disorder. Aims: The aim of this systematic literature review and meta‐analysis was to systematically examine the effects of different intervention approaches on speech production accuracy and phonological representation skills in children with speech and language disorders. Methods: A preregistered systematic review (International Prospective Register of Systematic Reviews ID: CRD42017076075) adhering to Preferred Reporting Items for Systematic Reviews and Meta‐Analyses guidelines was completed. Seven electronic databases (PubMed, Web of Science, ERIC, PsychINFO, Cochrane Library, SCOPUS and Linguistics & Language Behavior Abstracts) were searched for studies related to oral language interventions with children with developmental speech and/or language disorder (mean age ranging from 3–18 years) published between January 2006 and August 2022. The included articles reported intervention studies with a group design in which speech production accuracy was the outcome measure. Studies were appraised using the Cochrane risk of bias tool, and individual effect sizes were calculated using standardised means differences when enough data was available. A meta‐analysis was conducted obtaining the average standardised mean difference d. Heterogeneity, influence of possible moderator variables and publication bias were explored. Results: The 23 studies that met the inclusion criteria presented low‐medium risk of bias. Nine effect sizes were obtained from seven of these studies that presented a pre‐post‐test with a control group design. Medium‐high average effect sizes were found in phonological accuracy. Heterogeneity was found between individual effect sizes. Significant moderator variables and publication bias were not detected. Conclusions: The results of this meta‐analysis indicate positive effects on speech production accuracy. Based on this review, further improvements in the quality of reporting for intervention research are required in developing the evidence base for practice. What this paper adds: What is already known on the subject: An increasing number of interventions is available for children and adolescents with developmental speech and/or language disorders. Previous reviews suggest relatively low levels of evidence of interventions having phonology as an outcome measure. What this paper adds to the existing knowledge: This review and meta‐analysis summarise the intervention evidence from a substantial body of group design studies, indicating positive results from a range of interventions with phonological outcomes. It highlights the need to systematically implement and replicate different intervention procedures to understand factors that will maximise positive outcomes and to grow the evidence base for best practice. What are the potential or actual clinical implications of this study?: Tentative evidence is emerging for the effectiveness of various approaches in enhancing speech production accuracy skills of children and adolescents with developmental speech and/or language disorder. [ABSTRACT FROM AUTHOR]

Background: Speech sound disorders (SSDs) in children are heterogeneous. Differentiating children with SSDs into distinct subtypes is important so that each child receives a treatment approach well suited to the particular difficulties they are experiencing. Aims: To study the distinct underlying processes that differentiate phonological processing, phonological planning or motor planning deficits. Method: The literature on the nature of SSDs is reviewed to reveal diagnostic signs at the level of distal causes, proximal factors and surface characteristics. Main Contribution: Subtypes of SSDs may be identified by linking the surface characteristics of the children's speech to underlying explanatory proximal factors. The proximal factors may be revealed by measures of speech perception skills, phonological memory and speech–motor control. The evidence suggests that consistent phonological disorder (CPD) can be identified by predictable patterns of speech error associated with speech perception errors. Inconsistent phonological disorder (IPD) is associated with a deficit in the selection and sequencing of phonemes, that is, revealed as within‐word inconsistency and poor phonological memory. The motor planning deficit that is specific to childhood apraxia of speech (CAS) is revealed by transcoding errors on the syllable repetition task and an inability to produce [pətəkə] accurately and rapidly. Conclusions & Implications: Children with SSDs form a heterogeneous population. Surface characteristics overlap considerably among those with severe disorders, but certain signs are unique to particular subtypes. Careful attention to underlying causal factors will support the accurate diagnosis and selection of personalized treatment options. WHAT THIS PAPER ADDS: What is already known on the subject: SSD in children are heterogenous, with numerous subtypes of primary SSD proposed. Diagnosing the specific subtype of SSD is important in order to assign the most efficacious treatment approach for each child. Identifying the distinct subtype for each child is difficult because the surface characteristics of certain subtypes overlap among categories (e.g., CPD or IPD; CAS). What this paper adds to the existing knowledge: The diagnostic challenge might be eased by systematic attention to explanatory factors in relation to the surface characteristics, using specific tests for this purpose. Word identification tasks tap speech perception skills; repetition of short versus long strings of nonsense syllables permits observation of phonological memory and phonological planning skills; and standard maximum performance tests provide considerable information about speech motor control. What are the potential or actual clinical implications of this work?: Children with SSDs should receive comprehensive assessments of their phonological processing, phonological planning and motor planning skills frequently, alongside examinations of their error patterns in connected speech. Such assessments will serve to identify the child's primary challenges currently and as they change over developmental time. [ABSTRACT FROM AUTHOR]

Background: The publication of phase 2 of the CATALISE project in 2017 clarified terminology for children with developmental language disorder (DLD) or delay but unintentionally muddied the water for children with unintelligible speech. A diagnostic label of DLD (phonology) indicates poor prognosis and phonological disorder that persists into middle childhood. However, in contrast to other diagnostic labels that fall under the overarching term of speech sound disorder (SSD), DLD (phonology) does not elucidate the characteristics of the child's speech nor does it point us in the direction of appropriate intervention. Aims: The aim of this paper is to discuss terminology in SSD leading to an evidence‐based model which builds on the model of DLD developed in CATALISE, supports descriptive diagnosis and signposts intervention. Methods: Following a focused review of literature proposing or describing terminology for SSD, an expert group of researchers in developmental SSD proposed a revised model of existing terminology. Groups of UK speech and language therapists (SLTs) who provide services for children with SSD were asked to comment on its acceptability and feasibility. Discussion: A three‐level terminology model was developed. This comprised an overarching Level 1 term; Level 2 terms that differentiated SSD of unknown origin from SSD with associated or underlying conditions; and specific diagnostic terms at Level 3 to support further assessment and intervention decisions. Consulted SLTs generally expressed agreement with the proposed terminology and a willingness to adopt it in practice. Conclusions: Existing terminology for childhood SSD provides a good basis for clinical decision‐making. A modified version of Dodd's (2005) terminology was found to be acceptable to UK SLTs. There is an evident overlap of SSD with CATALISE terminology. However more detailed and specialist terminology than 'DLD (phonology)' is required to support clinical decision‐making. It is proposed that endorsement by the UK Royal College of Speech and Language Therapists would obviate the need for a Delphi process. What this paper adds: What is already known on this subject: Over nearly a hundred years, as our knowledge and understanding of speech sound disorder (SSD) has increased, so has the terminology that is used to describe those disorders. Current terminology not only describes subtypes of SSD but can also signpost us to effective interventions. With the publication, in 2017, of phase 2 of CATALISE a new term of 'developmental language disorder (DLD) (phonology)' was introduced with the unintentional consequence of challenging more specific descriptive terms for SSD. What this paper adds: In the context of CATALISE and DLD (phonology), the history and nature of SSD terminology are reappraised. Building on the model of DLD developed in CATALISE, a tiered model that supports descriptive diagnosis and signposts intervention is proposed for discussion. Clinical implications of this study: The proposed model of terminology for SSD provides descriptive and detailed labels that will support accuracy in differential diagnosis of developmental SSD by speech and language therapists. Furthermore, a decision‐making tree for SSD demonstrates the pathway from diagnostic use of the terminology to the selection of evidence‐based, effective interventions. [ABSTRACT FROM AUTHOR]

Background: 'Speech sound disorder' is an umbrella term that encompasses dysarthria, articulation disorders, childhood apraxia of speech and phonological disorders. However, differential diagnosis between these disorders is a persistent challenge in speech pathology, as many diagnostic procedures use symptom clusters instead of identifying an origin of breakdown in the speech and language system. Aims: This article reviews typical and disordered speech through the lens of two well‐developed models of production—one focused on phonological encoding and one focused on speech motor planning. We illustrate potential breakdown locations within these models that may relate to childhood apraxia of speech and phonological disorders. Main Contribution: This paper presents an overview of an approach to conceptualisation of speech sound disorders that is grounded in current models of speech production and emphasises consideration of underlying processes. The paper also sketches a research agenda for the development of valid, reliable and clinically feasible assessment protocols for children with speech sound disorders. Conclusion: The process‐oriented approach outlined here is in the early stages of development but holds promise for developing a more detailed and comprehensive understanding of, and assessment protocols for speech sound disorders that go beyond broad diagnostic labels based on error analysis. Directions for future research are discussed. What this paper adds: What is already known on the subject: Speech sound disorders (SSD) are heterogeneous, and there is agreement that some children have a phonological impairment (phonological disorders, PD) whereas others have an impairment of speech motor planning (childhood apraxia of speech, CAS). There is also recognition that speech production involves multiple processes, and several approaches to the assessment and diagnosis of SSD have been proposed. What this paper adds to existing knowledge: This paper provides a more detailed conceptualisation of potential impairments in children with SSD that is grounded in current models of speech production and encourages greater consideration of underlying processes. The paper illustrates this approach and provides guidance for further development. One consequence of this perspective is the notion that broad diagnostic category labels (PD, CAS) may each comprise different subtypes or profiles depending on the processes that are affected. What are the potential or actual clinical implications of this work?: Although the approach is in the early stages of development and no comprehensive validated set of tasks and measures is available to assess all processes, clinicians may find the conceptualisation of different underlying processes and the notion of potential subtypes within PD and CAS informative when evaluating SSD. In addition, this perspective discourages either/or thinking (PD or CAS) and instead encourages consideration of the possibility that children may have different combinations of impairments at different processing stages. [ABSTRACT FROM AUTHOR]

Purpose: The current study is a Phase I clinical study with the goal of determining feasibility and the effectiveness of the Kaufman Speech to Language Protocol (K-SLP) for children with childhood apraxia of speech (CAS) and comorbidities. We hypothesized that K-SLP intervention would result in improved outcomes and maintenance of treatment effect at 3–4 months postintervention. Method: Single-subject experimental design with multiple baselines across behaviors was replicated across a group of six children. Five out of six participants completed the study. The K-SLP intervention was administered in dyads four times a week for three consecutive weeks. Outcomes included assessment of word/syllable shapes, articulation accuracy, speech intelligibility, and functional communication. Treatment progress was measured through: (a) the administration of custom probe word lists and (b) assessments carried out at pretreatment, immediately following intervention and approximately 3–4 months after the study period. Results: Four out of five participants demonstrated significant improvements to words targeted in treatment and three out of five generalized these to untreated words. Furthermore, three out of five participants showed immediate and clinically significant posttreatment improvements in speech intelligibility and functional outcomes, and this increased to four out of five participants at 3–4 months follow-up. Conclusions: The study provides preliminary support for the effectiveness of the K-SLP program when delivered in dyads to children with CAS with comorbidities. The study replicates earlier findings and reaffirms the positive outcomes of K-SLP for children with CAS. [ABSTRACT FROM AUTHOR]

Purpose: The purpose of this investigation was to examine single-word speech intelligibility outcomes following sound production treatment in a group of 22 speakers with chronic acquired apraxia of speech (AOS) and aphasia. Also, the stability of repeated posttreatment intelligibility measures was examined for two scoring methods. Method: The Assessment of Intelligibility of Dysarthric Speech was administered twice to each participant at pretreatment and twice at 8 weeks posttreatment. The test–retest reliability of the pretreatment samples was evaluated in a prior study; repeated samples were found to be stable over sampling times. For the current study, the three expert listeners who had rated the pretreatment samples scored the posttreatment samples using transcription and multiplechoice scoring formats. An additional expert listener, blinded to sampling time, scored pre- and posttreatment samples. Results: The posttreatment samples were found to be stable over sampling times for the group. Posttreatment intelligibility scores were statistically significantly higher than the pretreatment scores for both scoring methods (i.e., increases of 9%–10%). Scores derived from the two scoring methods were strongly, positively correlated, with multiple-choice scores being significantly higher than transcription. The scoring methods did not differ significantly in the amount of change found from pre- to posttreatment. There were no statistically significant correlations between AOS severity and changes in pre- to posttreatment intelligibility scores. Performance for most participants was similar to group performance. Conclusions: Stability of posttreatment intelligibility samples supports use of intelligibility scores as outcome measures. This group of speakers demonstrated statistically significant increases in single-word speech intelligibility following sound production treatment. [ABSTRACT FROM AUTHOR]

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Senescence, marked by permanent cell cycle arrest may contribute to the decline in regenerative potential and neuronal function, thereby promoting neurodegenerative disorders. In this study, we employed whole exome sequencing to identify a previously unreported biallelic missense variant in SVBP (p.Leu49Pro) in six patients from three unrelated families. These affected individuals present with a complex hereditary spastic paraplegia (HSP), peripheral neuropathy, verbal apraxia, and intellectual disability, exhibiting a milder phenotype compared to patients with nonsense SVBP mutations described previously. Consistent with SVBP's primary role as a chaperone necessary for VASH‐mediated tubulin detyrosination, both patient fibroblasts with the p.Leu49Pro mutation, and HeLa cells harboring an SVBP knockdown exhibit microtubule dynamic instability and alterations in pericentriolar material (PCM) component trafficking and centrosome cohesion. In patient fibroblasts, structural abnormalities in the centrosome trigger mitotic errors and cellular senescence. Notably, premature senescence characterized by elevated levels of p16INK4, was also observed in patient peripheral blood mononuclear cells (PBMCs). Taken together, our findings underscore the critical role of SVBP in the development and maintenance of the central nervous system, providing novel insights associating cytokinesis failure with cortical motor neuron disease and intellectual disability. [ABSTRACT FROM AUTHOR]

Childhood apraxia of speech (CAS) is a severe and rare form of speech sound disorder (SSD). CAS is typically sporadic, but may segregate in families with broader speech and language deficits. We hypothesize that genetic changes may be involved in the etiology of CAS. We conduct whole-genome sequencing in 27 families with CAS, 101 individuals in all. We identify 17 genomic regions including 19 unique copy number variants (CNVs). Three variants are shared across families, but the rest are unique; three events are de novo. In four families, siblings with milder phenotypes co-inherited the same CNVs, demonstrating variable expressivity. We independently validate eight CNVs using microarray technology and find many of these CNVs are present in children with milder forms of SSD. Bioinformatic investigation reveal four CNVs with substantial functional consequences (cytobands 2q24.3, 6p12.3-6p12.2, 11q23.2-11q23.3, and 16p11.2). These discoveries show that CNVs are a heterogeneous, but prevalent, cause of CAS. Copy number variations identified in families with childhood apraxia of speech and other speech sound disorders provide a deeper understanding of the genetic basis for naturally acquired speech. [ABSTRACT FROM AUTHOR]

Purpose: Neurogenic speech and language disorders--such as acquired apraxia of speech (AOS) and aphasia with phonemic paraphasia (APP)--are often misdiagnosed due to similarities in clinical presentation. Word syllable duration (WSD)--a measure of average syllable length in multisyllabic words--serves as a proxy for speech rate, which is an important and arguably more objective clinical characteristic of AOS and APP. This study reports stability of WSD over time for speakers with AOS (and aphasia). Method: Twenty-nine participants with AOS and aphasia (11 women and 18 men, Mage = 53.5 years, SD = 13.3) repeated 30 multisyllabic words (of three-, four-, and five-syllable lengths) on three occasions across 4 weeks. WSDs were calculated for each word and then averaged across each list (i.e., word length), as well as across combined lists (i.e., all 30 words) to yield four WSDs for each participant at each time point. Stability over time was calculated using Friedman's test for the group and using Spearman's rho for the individual level. Effects of time and word length were examined using robust mixed-effects linear regression. Results: Friedman's tests and correlations indicated no significant difference in WSDs across sampling occasions for each word length separately or combined. WSD correlated positively with AOS severity and negatively with intelligibility but was not correlated with aphasia severity. Regression analyses confirmed WSD to be stable over time, while WSD calculated from only five tokens (i.e., WSD-5) was less stable over time. Conclusions: Results indicate that WSD can be a stable measure over time, at the individual and group level, providing support for its use in diagnosis and/or as an outcome measure, both clinically and for research. In general, WSD outperformed WSD-5, suggesting that it may be better to calculate WSD from more than five tokens. Stability of WSD in other populations and suitability for differential diagnosis need to be determined. Currently, differentiating disorders by speaking rate, alone, is not recommended. [ABSTRACT FROM AUTHOR]

Purpose: The purpose of this study was to investigate acoustic spatiotemporal variability in children with childhood apraxia of speech (CAS) compared to children with typical development (TD). Increased spatiotemporal variability in children with CAS relative to nonapraxic peers has been documented in multiple kinematic studies. To date, few studies have investigated spatiotemporal variability in CAS using an acoustic signal. Method: Data were drawn from 10 children with CAS and 10 children with TD, ranging in age from 5;0 to 11;2 (years;months), who participated in an online study. All children with CAS had a verified diagnosis and showed at least five CAS features across at least two tasks, independently confirmed by two speech-language pathologists with expertise in CAS. Children repeated the sentences "Buy Bobby a puppy" (BBAP) and "Mom pets the puppy" (MPP) 10 times each. The acoustic spatiotemporal index (STI), calculated from the amplitude envelope, was used to investigate acoustic spatiotemporal variability. Using a regression analysis, we analyzed group differences in STI values while controlling for age and gender. Results: Children with CAS did not produce enough usable tokens of BBAP to analyze. MPP revealed significantly higher acoustic STI values in children with CAS compared to children with TD. No significant effects were observed for age or gender. Acoustic data collected online without the use of specialized equipment yielded high-quality audio data from which amplitude envelope tracing could be reliably accomplished. Conclusions: This study adds to a growing body of empirical data indicating that children with CAS produce speech with more variable spatiotemporal control compared to children with TD. In addition to producing more variable speech, children with CAS were less consistently able to form productions free of phonetic errors. These findings also demonstrate the feasibility of using remote data collection to investigate acoustic spatiotemporal variability, which allows for the recruitment of larger samples of low-incidence populations. [ABSTRACT FROM AUTHOR]

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Purpose: We provide a case report of "Zoe," a 4-year-old deaf child from a deaf signing family, who presented with a possible case of Childhood Apraxia of Sign (CASign). Method: The description is based on reports from the child's speech-language pathologist, her Individualized Education Program report, and a clinician-created sign imitation task. Results: Zoe's sign articulation errors in American Sign Language differed from those reported for typically developing deaf children and were parallel to the types of errors observed for hearing children with childhood apraxia of speech. Specifically, Zoe produced inconsistent errors across signs, substituted more complex handshapes for simple handshapes, made errors on both unmarked (common) and marked (less common) forms, produced articulatory distortions (rather than substitutions), and exhibited "groping" behaviors (a sequence of attempts to move her hands into the correct position). In addition, Zoe sometimes self-corrected her errors by manipulating her own hands, for example, using her left hand to move the thumb of her right hand into the correct position. Conclusion: Zoe's pattern of sign errors is consistent with an underlying deficit in motor planning and/or programming and may constitute the first reported case of CASign. [ABSTRACT FROM AUTHOR]

Purpose: There is a need for stimuli that can be used in the assessment of apraxia of speech (AOS) characteristics. AOS often presents with aphasia, and the Western Aphasia Battery--Revised (WAB-R) is administered extensively in clinical practice for the assessment of aphasia. This preliminary study investigated error production of differentially diagnostic characteristics of AOS (phonetic and prosodic errors) in response to existing spoken language subtest stimuli of the WAB-R in individuals with concomitant AOS and aphasia and aphasia only. Method: Ten participants presenting with aphasia and potential motor speech impairment were included. Presence of AOS was determined for five individuals. Speech production errors were analyzed using narrow transcription methods in response to the Naming, Repetition, and Spontaneous Speech subtests of the WAB-R for both AOS and aphasia and aphasia-only groups. Results: Differences in total error production for both phonetic and prosodic errors were reported between the AOS and aphasia and aphasia-only groups. In addition, the phonetic complexity of the stimuli correlated to the phonetic and prosodic error production in both groups. An effect of WAB-R subtest was not reported for either group. Conclusion: The diversity of elicited spoken language production across multiple contexts makes the use of the spoken language subtest stimuli a feasible component of a comprehensive AOS assessment, since AOS is often concomitant with aphasia. [ABSTRACT FROM AUTHOR]

Purpose: Limited research exists assessing speech perception in school-age children with speech sound disorder (SSD) and childhood apraxia of speech (CAS); despite early evidence that speech perception may lead to error-prone motor planning/programming. In this study, we examine speech perception performance in school-age children with and without speech production deficits. Method: Speech perception was assessed using the Wide Range Acoustic Accuracy Scale to determine the just-noticeable difference in discrimination for three consonant-vowel syllable contrasts (/bɑ/-/wɑ/, /dɑ/-/gɑ/, /ɹɑ/-/wɑ/), each varying along a single acoustic parameter for seven children with CAS with rhotic errors, seven children with SSD with rhotic errors, and seven typically developing (TD) children. Results: Findings revealed statistically significant mean differences between perceptual performance of children with CAS when compared to TD children for discrimination of /ɹɑ/-/wɑ/ contrasts. Large effect sizes were also observed for comparisons of /ɹɑ/-/wɑ/ contrasts between children with CAS, SSD, and TD peers. Additionally, large effect sizes were observed for /dɑ/-/gɑ/ contrasts between children with CAS and SSD and TD children despite nonsignificant mean differences in group performance. Conclusions: Overall, mean outcome scores suggest that school-age children with CAS and persistent rhotic errors demonstrated less accurate speech perception skills relative to TD children for the /ɹɑ/-/wɑ/ contrasts. However, the relatively small sample sizes per group limit the extent to which these findings may be generalized to the broader population. [ABSTRACT FROM AUTHOR]

Purpose: The study's primary aims were to describe the long-term speech outcomes for adolescents and young adults with a history of childhood apraxia of speech (CAS) and to examine the association of persistent speech sound errors with measures of literacy skills, phonological processing, motor speech production, and parent report of early motor difficulty. Method: Data from a large longitudinal 25-year study were used to explore outcomes for 32 individuals with a history of CAS, ages 12;6 (years; months) to 25 years (M = 17.4, SD = 4.7). Persistent and nonpersistent groups were compared on decoding, phonological processing, multisyllabic word repetition, diadochokinetic rate, and parent report of motor involvement. Parametric (Welch's t tests) and nonparametric tests (Wilcoxon and Fisher exact tests) were used to identify differences between the groups' distributions. Developmental trajectories of speech production were plotted. Results: Outcomes for individuals with CAS are highly variable, with some demonstrating speech sound errors into adolescence and young adulthood. Speech sound errors were primarily on later developing sounds. Persistence was significantly associated with early motor difficulties. Difficulties with multisyllabic words, phonological processing, and literacy were often present regardless of persistence or nonpersistence of speech errors. Conclusions: Children with CAS are at risk for persistent speech sound errors into adulthood. For children showing limited progress with more traditional speech therapy, alternative interventions should be explored. Individuals with persistent speech sound errors are more likely to have a history of early motor deficits. Regardless of persistence, participants with CAS demonstrated ongoing weaknesses in literacy, phonological processing skills, and complex speech production tasks. [ABSTRACT FROM AUTHOR]

Purpose: The aim of this study was to explore relations between speech sound disorder severity and selective mutism in a group of children with 7q11.23 duplication syndrome (Dup7), a genetic condition predisposing children to childhood apraxia of speech (CAS) and other speech sound disorders and to anxiety disorders, including selective mutism and social anxiety disorder. Method: Forty-nine children aged 4-17 years with genetically confirmed Dup7 completed the Goldman-Fristoe Test of Articulation-Second Edition (GFTA-2), the Expressive Vocabulary Test-Second Edition (EVT-2), and the Differential Ability Scales-Second Edition (DAS-II). Parents completed the Anxiety Disorders Interview Schedule-Parent (ADIS-P). Results: Mean standard scores (SSs) were 65.67 for the GFTA-2, 92.73 for the EVT-2, and 82.69 for the DAS-II General Conceptual Ability (GCA; similar to IQ). Standard deviations for all measures were larger than for the general population. GFTA-2 SS was significantly correlated with both EVT-2 SS and DAS-II GCA. Based on the ADIS-P, 22 participants (45%) were diagnosed with selective mutism and 29 (59%) were diagnosed with social anxiety disorder. No significant differences in performance on any of the measures were found either between the group with a selective mutism diagnosis and the group that did not have selective mutism or between the group with a selective mutism and/or social anxiety disorder diagnosis and the group that did not have either disorder. Conclusions: For children with Dup7, neither the diagnosis of selective mutism nor the diagnosis of selective mutism and/or social anxiety disorder was related to severity of speech sound disorder, expressive vocabulary ability, or overall intellectual ability. Accordingly, treatment for speech sound disorder alone is unlikely to lead to remission of selective mutism or social anxiety disorder. Instead, selective mutism and/or social anxiety disorder should be treated directly. Further research is needed to determine if these findings generalize to other populations, such as children with idiopathic CAS. [ABSTRACT FROM AUTHOR]

Background: Speech and language are interconnected systems, and language disorder often co-occurs with childhood apraxia of speech (CAS) and non-CAS speech sound disorders (SSDs). Potential trade-off effects between speech and language in connected speech in children without overt language disorder have been less explored. Method: Story retell narratives from 24 children (aged 5;0-6;11 [years;months]) with CAS, non-CAS SSD, and typical development were analyzed in Systematic Analysis of Language Transcripts (SALT) regarding morphosyntactic complexity (mean length of C-unit in words [MLCU]), lexical diversity (moving-average type-token ratio [MATTR]), and linguistic accuracy (any linguistic error/bound morpheme omissions) and compared to 128 age-matched children from the SALT database. Linear and mixed-effects logistic regressions were performed with speech accuracy (percent phonemes correct [PPC]) and diagnostic group as predictors of the narrative variables. Results: PPC predicted all narrative variables. Poorer PPC was associated with lower MLCU and MATTR as well as a higher likelihood of linguistic errors. Group differences were only observed for the error variables. Comparison to the SALT database indicated that 13 of 16 children with CAS and SSD showed a higher-than-expected proportion of linguistic errors, with a small proportion explained by individual speech errors only. Conclusions: The high occurrence of linguistic errors, combined with the relationship between PPC and linguistic errors in children with CAS/SSD, suggests a trade-off between speech accuracy and language output. Longitudinal studies are needed to investigate whether children with SSDs without language disorder show more language difficulties over time as linguistic demands increase. [ABSTRACT FROM AUTHOR]

Purpose: This study examines how ultrasound biofeedback and intensive treatment distribution affect speech sound generalization during an evidence-based treatment, Speech Motor Chaining, for children with persisting speech errors associated with childhood apraxia of speech (CAS). Method: In a 2 x 2 factorial randomized controlled trial, children ages 9-17 years meeting CAS criteria were randomized to receive (a) a distributed treatment (20 sessions twice weekly over 10 weeks) or intensive treatment (20 hr in 5 weeks, with 10 hr in Week 1) and (b) treatment with or without biofeedback. Due to the COVID pandemic, some participants were randomized to distributed/intensive telepractice treatment only. The primary outcome was percent target sounds correct on untreated phrases (i.e., generalization) at the 10-week time point. More than 50,000 narrow phonetic transcriptions were analyzed. Results: Forty-eight participants completed treatment. Intensive treatment significantly increased generalization at all time points. The effect of biofeedback was significant at 5 weeks from the start of treatment but not significant at the primary 10-week time point. However, when comparing each group immediately after their 20 hr of treatment finished, generalization was significantly greater in intensive over distributed treatment and greater in ultrasound over no-ultrasound treatment (with a significant interaction favoring intensive treatment with ultrasound). Only the advantage of intensive treatment remained significant 5 weeks after groups finished treatment. There was no significant difference between face-to-face and telepractice modalities. Conclusions: When the number of treatment hours is fixed, an intensive schedule of Speech Motor Chaining facilitated greater improvement than a distributed schedule. Ultrasound biofeedback initially accelerated learning, but the benefits may dissipate as treatment continues or after it ends. [ABSTRACT FROM AUTHOR]

Purpose: The aim of this study was to pilot the efficacy of rapid syllable transition (ReST) treatment when provided once per week for a 50-min treatment session for 12 weeks with five children with childhood apraxia of speech. Of central importance was the children's retention and generalization of gains from treatment as indicators of speech motor learning. Method: A multiple-baseline across-participant design was employed to investigate (a) treatment effect on the 20 treated pseudowords, (b) generalization to 40 untreated real words and 10 untreated polysyllabic word sentences, and (c) maintenance of any treatment and generalization goals to up to 4 months posttreatment. To investigate any difference between in-session performance and retention, a comparison was made between data collected during treatment and probe sessions. Results: Treatment data collected during therapy showed all children improving across their 12 treatment sessions. Three of the five children showed a treatment effect on treated pseudowords in the probe sessions, but only one child showed generalization to untreated real words, and no children showed generalization to sentences. Conclusions: ReST treatment delivered at a dose frequency of once per week was efficacious for only one of the five children. In-session treatment data were not a reliable indicator of children's learning. One session per week of ReST therapy is therefore not recommended. Supplemental Material: https://doi.org/10.23641/asha.23751018 [ABSTRACT FROM AUTHOR]

Purpose: The aim of this study was to describe changes in speech intelligibility and interrater and intrarater reliability of naive listeners' ratings of words produced by young children diagnosed with childhood apraxia of speech (CAS) over a period of motor-based intervention (dynamic temporal and tactile cueing [DTTC]). Method: A total of 120 naive listeners (i.e., listeners without experience listening to children with speech and/or language impairments; age range: 18-45 years) orthographically transcribed single-word productions by five children (age range: 2;6-3;11 [years;months]) across three time points over an intervention period (baseline, post-treatment, maintenance). Changes in intelligibility and interrater and intrarater reliability were examined within and across time points. Results: Speech intelligibility significantly increased in children with CAS over the course of treatment, and these gains were also maintained at 6 weeks posttreatment. There was poor-to-fair consistency between listeners (interrater reliability) and excellent consistency within listeners (intrarater reliability) in ratings of speech intelligibility within and across time points. Conclusions: Motor-based intervention increases speech intelligibility following a period of DTTC treatment. Variability among naive listeners of speech intelligibility was also present, with intrarater reliability (within listeners) yielding greater consistency than interrater reliability (between listeners). The implications for including naive listeners as raters of speech intelligibility for research and clinical purposes are discussed. [ABSTRACT FROM AUTHOR]

Purpose: Speech motor skill is refined over the course of practice, which is commonly reflected by increased accuracy and consistency. This research examined the relationship between auditory-perceptual ratings of word accuracy and measures of speech motor timing and variability at pre- and posttreatment in children with childhood apraxia of speech (CAS). Furthermore, the degree to which individual patterns of baseline probe word accuracy, receptive language, and cognition predicted response to treatment was explored. Method: Probe data were collected from seven children with CAS (aged 2;5-5;0 [years;months]) who received 6 weeks of Dynamic Temporal and Tactile Cueing (DTTC) treatment. Using a multidimensional approach to measuring speech performance, auditory-perceptual (whole-word accuracy), acoustic (whole-word duration), and kinematic (jaw movement variability) analyses were conducted on probe words produced pre- and posttreatment. Standardized tests of receptive language and cognition were administered pretreatment. Results: There was a negative relationship between auditory-perceptual measures of word accuracy and movement variability. Higher word accuracy was associated with lower jaw movement variability following intervention. There was a strong relationship between word accuracy and word duration at baseline, which became less robust posttreatment. Furthermore, baseline word accuracy was the only child-specific factor to predict response to DTTC treatment. Conclusions: Following a period of motor-based intervention, children with CAS appeared to refine speech motor control in conjunction with improvements in word accuracy. Those who demonstrated the poorest performance at treatment onset displayed the greatest degree of gains. Taken together, these results reflect a system-wide change following motor-based intervention. [ABSTRACT FROM AUTHOR]

Purpose: Advancements in genetic testing and analysis have allowed improved identification of the genetic basis of childhood apraxia of speech, a rare speech presentation. This study aimed to understand speech-language pathologists' (SLPs') consideration of incorporation of genetics in clinical practice using a theory-informed qualitative approach. Method: Semistructured interviews were conducted with 12 pediatric SLPs using a behavior change theory (Theoretical Domains Framework [TDF]) within a case study describing a child with complex co-occurring features, including childhood apraxia of speech. Interviews focused on three stages of the patient journey (prereferral, referral, and postreferral). Interviews were analyzed to identify barriers and enablers to considering incorporation of genetics in current clinical practice. Barriers and enablers were grouped and mapped onto a contextually relevant TDF-coded analysis framework. Results: Barriers were identified across several TDF domains, through all stages of the patient journey. Lack of confidence, relevance, and level of experience were most common prereferral, and connection to and awareness of genetics services and contextual factors were barriers in the referral stage. Perception of professional role, knowledge, and beliefs about effects on families were barriers postreferral. Associated enablers were also identified, including seeing value in genetic diagnosis, support from other health care professionals, supervision, and relationships with genetics services. Conclusions: Results of this qualitative study highlight barriers and enablers to incorporating genetics into speech-language pathology clinical practice. These findings will assist in the development of theory-informed implementation strategies to support SLPs into the future. [ABSTRACT FROM AUTHOR]

Purpose: The current standard for clinical diagnosis of childhood apraxia of speech (CAS) is expert clinician judgment. The psychometric properties of this standard are not well understood; however, they are important for improving clinical diagnosis. The purpose of this study is to determine the extent to which experts agree on the clinical diagnosis of CAS using two cohorts of children with mixed speech sound disorders (SSDs). Method: Speech samples of children with SSDs were obtained from previous and ongoing research from video recordings of children aged 3-8 years (n = 36) and audio recordings of children aged 8-17 years (n = 56). A total of 23 expert, English-speaking clinicians were recruited internationally. Three of these experts rated each speech sample to provide a description of the observed features and a diagnosis. Intrarater reliability was acceptable at 85% agreement. Results: Interrater reliability on the presence or absence of CAS among experts was poor both as a categorical diagnosis (κ = .187, 95% confidence interval [CI] [0.089, 0.286]) and on a continuous "likelihood of CAS" scale (0-100; intraclass correlation = .183, 95% CI [.037, .347]). Reliability was similar across the video-recorded and audio-only samples. There was greater agreement on other diagnoses (such as articulation disorder) than on the diagnosis of CAS, although these too did not meet the predetermined standard. Likelihood of CAS was greater in children who presented with more American Speech-Language-Hearing Association CAS consensus features. Conclusions: Different expert raters had different thresholds for applying the diagnosis of CAS. If expert clinician judgment is to be used for diagnosis of CAS or other SSDs, further standardization and calibration is needed to increase interrater reliability. Diagnosis may require operationalized checklists or reliable measures that operate along a diagnostic continuum. Supplemental Material: https://doi.org/10.23641/asha.23949105 [ABSTRACT FROM AUTHOR]

Purpose: The purpose of this tutorial is to (a) provide an updated review of the literature pertaining to proposed early features of childhood apraxia of speech (CAS), (b) discuss the findings of recent treatment studies of infants and toddlers with suspected CAS (sCAS), and (c) present evidence-based strategies and tools that can be used for the identification of and intervention for infants and toddlers with sCAS or at high risk for the disorder. Method: Since Davis and Velleman's (2000) seminal work on assessment and intervention in infants and toddlers with sCAS, limited research has guided clinicians in the complex task of identifying and treating early speech motor difficulties prior to a definitive diagnosis of CAS. Following the structure of Davis and Velleman, we explore the proposed early characteristics of CAS with reference to contemporary research. Next, we describe the limited treatment studies that have investigated intervention for infants and toddlers at risk of or suspected of having CAS. Finally, we present practical suggestions for integrating this knowledge into clinical practice. Conclusions: Many of the originally proposed correlates of CAS in infants and toddlers now have research supporting their presence. However, questions remain about the developmental trajectory of the disorder. Although limited in number and restricted by lack of experimental control, emerging treatment studies can help guide clinicians in providing appropriate intervention to infants and toddlers with sCAS who need not wait for a definitive diagnosis to initiate intervention. [ABSTRACT FROM AUTHOR]

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. [ABSTRACT FROM AUTHOR]

This article introduces the Journal of Speech, Language, and Hearing Research Special Issue: Selected Papers From the 2022 Apraxia Kids Research Symposium. The field of childhood apraxia of speech (CAS) has developed significantly in the past 15 years, with key improvements in understanding of basic biology including genetics, neuroscience, and computational modelling; development of diagnostic tools and methods; diversity of evidence-based interventions with increasingly rigorous experimental designs; and understanding of impacts beyond impairment-level measures. Papers in this special issue not only review and synthesize the some of the substantial progress to date but also present novel findings addressing critical research gaps and adding to the overall body of knowledge. A second aim of this prologue is to report the current research needs in CAS, which arose from symposium discussions involving researchers, clinicians, and Apraxia Kids community members (including parents of children with CAS). Four primary areas of need emerged from discussions at the symposium. These were: (a) What questions should we ask? (b) Who should be in the research? (c) How do we conduct the research? and (d) How do we move from research to practice? Across themes, symposium attendees emphasized the need for CAS research to better account for the diversity of people with CAS and improve the timeliness of implementation of high-level evidence-based practice across the lifespan. It is our goal that the articles and prologue discussion in this special issue provide an appreciation of advancements in CAS research and an updated view of the most pressing needs for future research. [ABSTRACT FROM AUTHOR]

Primary progressive aphasia (PPA) and primary progressive apraxia of speech (PPAOS) are neurodegenerative syndromes characterized by progressive decline in language or speech. There is a growing number of studies investigating speech-language interventions for PPA/PPAOS. An updated systematic evaluation of the treatment evidence is warranted to inform best clinical practice and guide future treatment research. We systematically reviewed the evidence for behavioral treatment for speech and language in this population. Reviewed articles were published in peer-reviewed journals through 31 May 2021. We evaluated level of evidence, reporting quality, and risk of bias using a modified version of the American Speech-Language Hearing Association (ASHA) Levels of Evidence, an appraisal point system, additional reporting quality and internal/external validity items, and, as appropriate, the Single Case Experimental DesignScale or the Physiotherapy Evidence Database – PsycBITERating Scale for Randomized and Non-Randomized Controlled Trials. Results were synthesized using quantitative summaries and narrative review. A total of 103 studies reported treatment outcomes for 626 individuals with PPA; no studies used the diagnostic label PPAOS. Most studies evaluated interventions for word retrieval. The highest-quality evidence was provided by 45 experimental and quasi-experimental studies (16 controlled group studies, 29 single-subject designs). All (k = 45/45) reported improvement on a primary outcome measure; most reported generalization (k = 34/43), maintenance (k = 34/39), or social validity (k = 17/19) of treatment for at least one participant. The available evidence supports speech-language intervention for persons with PPA; however, treatment for PPAOS awaits systematic investigation. Implications and limitations of the evidence and the review are discussed. [ABSTRACT FROM AUTHOR]

Dysarthria and Apraxia of Speech (AoS) are motor speech disorders in which neurological lesions differentially affect motor control, possibly leading to noticeable differences in articulation and consequently sound production. Among the sounds requiring greater motor capacity because of its articulatory complexity is the voiceless alveolar sibilant fricative /s/. The aim of this study was to identify acoustic variables able to distinguish between dysarthria and AoS, and between these disorders and normal speech in Spanish speakers. The production of this fricative was acoustically examined in 28 individuals with motor neurological disorders (20 with dysarthria, 8 with AoS) and in 28 neurologically healthy persons. Participants repeated 12 monosyllabic words containing the fricative plus one of the five Spanish vowels. The variables measured were absolute durations of the fricative, vowel, and fricative+vowel sequence, along with the vowel-to-fricative duration ratio. Findings indicate that duration of the fricative can distinguish between controls and speakers with dysarthria, but not between controls and speakers with AoS. Measures related to vowel duration served to distinguish between speakers with dysarthria and speakers with AoS and between each of them and controls. Further, speakers with dysarthria and those with AoS differed from each other and from controls in terms of articulatory variability; speakers with dysarthria showing most variability. In the latter participants, articulatory variability was higher for unrounded segments, vowels and fricatives, while in speakers with AoS this variability was higher for rounded segments. These observations are discussed within a framework of motor control models. [ABSTRACT FROM AUTHOR]

Purpose: This study aimed to determine the outcomes and impact of Dynamic Temporal and Tactile Cueing (DTTC) treatment when clear vinyl masks were worn. DTTC is one of the few evidence-based treatments for children with childhood apraxia of speech (CAS). Given that DTTC relies on visual, auditory, and tactile cues, it was unknown if treatment gains would be demonstrated when masks were worn and how masking would impact the therapy experience for clinicians and caregivers. Method: A sequential mixed methods design was used to study the efficacy of DTTC treatment in children with CAS when clear masks were worn. The quantitative phase used a multiple-baseline across-participants design. Four children (each 4 years of age) participated in the treatment protocol in which 24 sessions of DTTC were provided over 8 weeks while clear vinyl face masks were worn by participants and clinicians. Whole word accuracy on treated items and generalization to easy and hard untreated items were assessed during baseline, treatment, and follow-up. Semistructured interviews were conducted with clinicians and caregivers following treatment to explore the experience of masks being worn during treatment. Qualitative data were analyzed using descriptive thematic analysis. Results: Three children completed the treatment protocol. Visual and statistical analyses revealed that two participants demonstrated significant treatment effects, with one also demonstrating generalization. The remaining participant demonstrated marginal treatment gains. Qualitative findings revealed two main themes: "mask wearing was inconvenient but did not prevent therapy gains" and "in-person therapy with face masks was preferable to teletherapy." Conclusions: Masks did not prohibit treatment gains during DTTC therapy, with treatment effects of varying degrees shown for the three participants who completed the protocol. Together, quantitative and qualitative results indicate that mask wearing was, for most, a minor inconvenience that did not substantially interfere with the efficacy of DTTC treatment. [ABSTRACT FROM AUTHOR]

Introduction: Transcribing disordered speech can be useful when diagnosing motor speech disorders such as primary progressive apraxia of speech (PPAOS), who have sound additions, deletions, and substitutions, or distortions and/or slow, segmented speech. Since transcribing speech can be a laborious process and requires an experienced listener, using automatic speech recognition (ASR) systems for diagnosis and treatment monitoring is appealing. This study evaluated the efficacy of a readily available ASR system (wav2vec 2.0) in transcribing speech of PPAOS patients to determine if the word error rate (WER) output by the ASR can differentiate between healthy speech and PPAOS and/or among its subtypes, whether WER correlates with AOS severity, and how the ASR's errors compare to those noted in manual transcriptions. Method: Forty-five patients with PPAOS and 22 healthy controls were recorded repeating 13 words, 3 times each, which were transcribed manually and using wav2vec 2.0. The WER and phonetic and prosodic speech errors were compared between groups, and ASR results were compared against manual transcriptions. Results: Mean overall WER was 0.88 for patients and 0.33 for controls. WER significantly correlated with AOS severity and accurately distinguished between patients and controls but not between AOS subtypes. The phonetic and prosodic errors from the ASR transcriptions were also unable to distinguish between subtypes, whereas errors calculated from human transcriptions were. There was poor agreement in the number of phonetic and prosodic errors between the ASR and human transcriptions. Conclusions: This study demonstrates that ASR can be useful in differentiating healthy from disordered speech and evaluating PPAOS severity but does not distinguish PPAOS subtypes. ASR transcriptions showed weak agreement with human transcriptions; thus, ASR may be a useful tool for the transcription of speech in PPAOS, but the research questions posed must be carefully considered within the context of its limitations. [ABSTRACT FROM AUTHOR]

We encountered two acute stroke cases with apraxia of speech (AOS) due to left internal border‐zone infarction. Neither case had a lower precentral gyrus lesion in the left frontal lobe. The findings obtained confirmed the relationship between AOS with improvement and damage to the left frontal aslant tract (FAT) in stroke. FAT integrity has potential as a prognostic imaging biomarker of AOS. It is important to visualize the fiber connections of white matter using diffusion tensor tractography, which will predict the outcome of AOS. [ABSTRACT FROM AUTHOR]

The cerebellum is traditionally known to subserve motor functions. However, for several decades, the concept of the "cerebellar cognitive affective syndrome" has evolved. Studies in healthy participants and patients have confirmed the cerebellar role in language. The exact involvement of the cerebellum regarding cerebellar aphasia remains uncertain. We included 43 cerebellar stroke patients who were tested at 3 months post-onset with the Boston Naming Test (BNT), the Token Test (TT), and the Diagnostic Instrument for Mild Aphasia (DIMA). Lesion side (left/right) and volume (cm3) were investigated. Patients significantly deviated on the following: BNT (p<0.001), TT (p<0.05), DIMA subtests: sentences repetition (p=0.001), semantic odd-picture-out (p<0.05), sentence completion (p<0.05) without an effect of lesion location (left/right) or volume (cm3) (p>0.05). Our clinical study confirms a non-lateralized cerebellar aphasia post-stroke, characterized by impairments in word retrieval, phonology, semantics, and syntax resembling cerebral-induced aphasia. The integral cerebellum appears to interact with eloquent cortico-subcortical language areas. [ABSTRACT FROM AUTHOR]

Ataxia-ocular apraxia 2 (AOA2) is a rare neurodegenerative autosomal recessive disorder with no effective treatment. In this study, we present the case of a patient diagnosed with AOA2, who experienced walking instability and uncoordinated movement. The patient underwent transcranial alternating current stimulation (tACS) treatment for 4 weeks with follow-up after 1 month. The effectiveness of the treatment was evaluated using the International Cooperative Ataxia Rating Scale (ICARS), the Scale for the Assessment and Rating of Ataxia (SARA), the 9-Hole Peg Test (9HPT), and functional near-infrared spectroscopy (fNIRS). Following treatment, the patient's ataxia symptoms showed significant improvement and continued to be alleviated during the follow-up period, suggesting a lasting effect of tACS treatment. Our findings from this case study provide compelling evidence for the potential of tACS as a treatment option for AOA2. [ABSTRACT FROM AUTHOR]

Speech is the most common means of communication in humans. Any defect in accurate speech production ability results in the development of speech sound disorder (SSD), a condition that can significantly impair an individual's academic performance, social interactions, and relationships with peers and adults. This study investigated the genetic basis of SSD in three Pakistani families. We performed family-based genome-wide parametric linkage analysis and homozygosity mapping in three consanguineous families with SSD from the Punjab province of Pakistan. The Test for Assessment of Articulation and Phonology in Urdu (TAAPU) was used to analyze the speech articulation data and determine the Percentage Correct Consonants (PCC) score. The PCC score defined the affected and unaffected individuals in each family. Parametric linkage analysis revealed a linkage to chromosome 5 (5q21.3-5q23.1) with a significant logarithm of the odds (LOD) score of 3.13 in a Pakistani family with specific language impairment-97 (PKSLI-97) under an autosomal recessive mode of inheritance. The other two families showed a suggestive linkage at 6p22.1, 14q12, and 16q12.1 under the recessive mode of inheritance. Interestingly, homozygosity mapping showed a loss of heterozygosity in the linkage region at 5q15-5q23.1, shared among seven affected (mostly in the younger generation) and one unaffected individual of PKSLI-97. Our analysis identified the 6p22 locus previously implicated in dyslexia, childhood apraxia of speech (CAS), and language impairment, confirming the role of KIAA0319 and DCDC2 in this locus. These findings provide statistical evidence for the genomic regions associated with articulation disorder and offer future opportunities to further the role of genes in speech production. [ABSTRACT FROM AUTHOR]

AbstractOverviewMethodResultConclusionNon-word repetition (NWR) is one of the most effective predictors of language impairments in children as it has been found to correlate with various language measures and the association between NWR and vocabulary is well documented in typically developing (TD) studies. However, there is a dire need for investigations of language skills in Kuwaiti Arabic individuals with Down Syndrome, and this study set out to fill a gap in this field.In this paper, we compare the vocabulary and NWR skills of a group of 48 individuals with DS aged 6–20 years to a group of 44 TD children aged 3–10 years matched on nonverbal IQ. Furthermore, we investigate the correlations among these language measures in the two groups and examine whether NWR can predict receptive and expressive vocabulary in these two groups.Results found DS participants performed significantly less than the TD group on the three language measures (receptive vocabulary t(90)= −3.17, p < .01, expressive vocabulary t(90)= −3.27, p < .01, and NWR t(90)= −8.32, p < .01). Moreover, there were strong correlations between NWR and vocabulary (receptive and expressive) in the TD group but not the DS group.Findings supported the working memory model and the phonological processing account for the TD group. On the other hand, the poor association between NWR and vocabulary in the DS group might be due to poor phonological discrimination difficulties and speech discrimination difficulties. [ABSTRACT FROM AUTHOR]

This study compared language samples from typically developing 4-year-olds who spoke African American English (AAE), Jamaican English (JE), or Mainstream American English (MAE) to assess the value of using language sample analysis (LSA) measures for characterising language use across dialects of English. Specific LSA metrics included mean length of utterance (MLU) in morphemes and in words, the Index of Productive Syntax (IPSyn), Developmental Sentence Scoring (DSS) and measures of lexical diversity. Children demonstrated diverse linguistic patterns across dialects, but a Kruskal–Wallis H test did not reveal significant differences in scores obtained through LSA measures. Notably, the IPSyn captured morphosyntactic structures in each category across dialects where prior research has highlighted limitations. This preliminary study uses a case-based approach to illustrate the applicability of LSAs in describing linguistic variations across children who speak different dialects of English. Moreover, the findings from this study underscore the potential use of LSAs in describing linguistic patterns to support the characterisation of communication profiles for culturally and linguistically diverse children. [ABSTRACT FROM AUTHOR]

BackgroundAimsMethods & ProceduresOutcomes & ResultsConclusionsSelf-advocacy scripts are brief, scripted narratives that people with aphasia (PWA) use to self-disclose their aphasia, explain what it is, and request supportive communicative behaviors from communication partners (CPs). In previous research, PWA have reported that self-advocacy statements, including self-advocacy scripts, facilitate their interactions with unfamiliar CPs. To understand the benefits of self-advocacy scripts, it is important to directly investigate how unfamiliar CPs respond to them.The aim of this study was to characterize the thoughts and emotions reported by unfamiliar adults after listening to self-advocacy scripts produced by speakers with Broca’s aphasia and apraxia of speech.This experimental study included 238 adult research participants without aphasia. Each participant was randomly assigned to listen to one script produced by a speaker with Broca’s aphasia and apraxia of speech: either a self-advocacy script or a control script about the weather that does not mention the speaker’s aphasia. Then, they answered one open-ended question (analyzed using a codebook approach) and eight rating scale questions (analyzed using Bayesian linear regression) about the thoughts and emotions they experienced while listening to the speaker.Compared to the control script, the self-advocacy script improved listeners’ knowledge about the speakers’ condition, their perception of the speakers’ intelligence, and their patience and empathy for the speakers.Aphasia self-advocacy scripts can have powerful, positive effects on unfamiliar listeners. The present results highlight the importance of providing support in clinical practice for collaborative development of self-advocacy scripts between people with aphasia and their clinicians. [ABSTRACT FROM AUTHOR]

Background: Traumatic brain injury (TBI), particularly damage to the frontal lobes, often produces a specific configuration of behavioral and personality changes known as frontal syndrome. Recent data, however, indicate that patients, depending on whether the damage has occurred in the left or right hemisphere of the brain, manifest one of two profiles of this syndrome in which negative or positive behavioral disturbances predominate. The purpose of this study was to evaluate the differences in frontal syndrome profiles present in patients 20 years after injury to the frontal lobes of the right and left hemispheres. Material/Methods: The study included a total of 360 patients with brain injury confirmed by neuroimaging studies, rehabilitated at the Reintegration and Training Center of the Polish Neuropsychological Society. The subjects were matched by age and gender into 2 experimental groups: Group A, included 180 patients (including 90 men and 90 women) with post-traumatic frontal lobe damage in the left hemisphere of the brain, and Group B, which included 180 patients (including 90 men and 90 women) with post-traumatic frontal lobe damage in the right hemisphere of the brain. The study employed documentation analysis, clinical interview and the Frontal Syndrome Behavior Questionnaire (FBInv). In accordance with the guidelines of this questionnaire, assessments were made based on an interview with the patient's caregiver in the absence of the patient. The study was conducted twice: two years (baseline) and about 20 years (follow up) after the brain injury. Results: It was found that thepatients with left frontal lobe damage were characterised by significantly higher levels of so-called negative behavioural disorders overall and in the dimensions of apathy, passivity, indifference, con cre teness, disorganisation, logopenia, apraxia of speech and perseveration, as well as significantly lower levels of loss of insight. While the patients with right frontal lobe damage were characterised by significantly higher levels of so-called positive conduct disorder overall and in the dimensions of hypersensitivity and irritability, excessive cheerfulness, unreasonable behaviour, inap propriateness and an absence of polite behaviour, aggressiveness, hy per-orality, hyper-sexuality, compulsivity, urinary and faecal incontinen ce and alien handedness. In addition, it should be noted that most of the recorded effects were found to be strong (η2> 0.14). The results indicated that 20 years after the brain injury, in terms of positive behavioural disorders overall, the group of patients with right frontal lobe damage showed a stronger decrease in disorders (η2= 0.64) over time when compared to patients with left frontal lobe damage (η2= 0.14). Conclusions: We found that in the patients 20 years after post-traumatic damage to the frontal lobes is still manifesting frontal syndrome, however its profile is dependent on whether the damage occurred in the left frontal lobe or the right frontal lobe. The Frontal Syndrome Behavior Questionnaire can be used in the diffe - rential diagnosis of frontal syndrome after damage to the frontal lobes of the right and left hemispheres of the brain. [ABSTRACT FROM AUTHOR]

Background: Dementia is a general term for several progressive neurodegenerative disorders including Alzheimer's disease. Timely and accurate detection is crucial for early intervention. Advancements in artificial intelligence present significant potential for using machine learning to aid in early detection. Objective: Summarize the state-of-the-art machine learning-based approaches for dementia prediction, focusing on non-invasive methods, as the burden on the patients is lower. Specifically, the analysis of gait and speech performance can offer insights into cognitive health through clinically cost-effective screening methods. Methods: A systematic literature review was conducted following the PRISMA protocol (Preferred Reporting Items for Systematic Reviews and Meta-Analyses). The search was performed on three electronic databases (Scopus, Web of Science, and PubMed) to identify the relevant studies published between 2017 to 2022. A total of 40 papers were selected for review. Results: The most common machine learning methods employed were support vector machine followed by deep learning. Studies suggested the use of multimodal approaches as they can provide comprehensive and better prediction performance. Deep learning application in gait studies is still in the early stages as few studies have applied it. Moreover, including features of whole body movement contribute to better classification accuracy. Regarding speech studies, the combination of different parameters (acoustic, linguistic, cognitive testing) produced better results. Conclusions: The review highlights the potential of machine learning, particularly non-invasive approaches, in the early prediction of dementia. The comparable prediction accuracies of manual and automatic speech analysis indicate an imminent fully automated approach for dementia detection. [ABSTRACT FROM AUTHOR]

Background and purpose: Logopenic variant primary progressive aphasia (lvPPA) is a major variant presentation of Alzheimer's disease (AD) that signals the importance of communication dysfunction across AD phenotypes. A clinical staging system is lacking for the evolution of AD‐associated communication difficulties that could guide diagnosis and care planning. Our aim was to create a symptom‐based staging scheme for lvPPA, identifying functional milestones relevant to the broader AD spectrum. Methods: An international lvPPA caregiver cohort was surveyed on symptom development under an 'exploratory' survey (34 UK caregivers). Feedback from this survey informed the development of a 'consolidation' survey (27 UK, 10 Australian caregivers) in which caregivers were presented with six provisional clinical stages and feedback was analysed using a mixed‐methods approach. Results: Six clinical stages were endorsed. Early symptoms included word‐finding difficulty, with loss of message comprehension and speech intelligibility signalling later‐stage progression. Additionally, problems with hearing in noise, memory and route‐finding were prominent early non‐verbal symptoms. 'Milestone' symptoms were identified that anticipate daily‐life functional transitions and care needs. Conclusions: This work introduces a new symptom‐based staging scheme for lvPPA, and highlights milestone symptoms that could inform future clinical scales for anticipating and managing communication dysfunction across the AD spectrum. [ABSTRACT FROM AUTHOR]

Background and objectives: Nonfluent variant primary progressive aphasia (nfvPPA) and primary progressive apraxia of speech (PPAOS) can be precursors to corticobasal syndrome (CBS). Details on their progression remain unclear. We aimed to examine the clinical and neuroimaging evolution of nfvPPA and PPAOS into CBS. Methods: We conducted a retrospective longitudinal study in 140 nfvPPA or PPAOS patients and applied the consensus criteria for possible and probable CBS for every visit, evaluating limb rigidity, akinesia, limb dystonia, myoclonus, ideomotor apraxia, alien limb phenomenon, and nonverbal oral apraxia (NVOA). Given the association of NVOA with AOS, we also modified the CBS criteria by excluding NVOA and assigned every patient to either a progressors or non-progressors group. We evaluated the frequency of every CBS feature by year from disease onset, and assessed gray and white matter volume loss using SPM12. Results: Asymmetric akinesia, NVOA, and limb apraxia were the most common CBS features that developed; while limb dystonia, myoclonus, and alien limb were rare. Eighty-two patients progressed to possible CBS; only four to probable CBS. nfvPPA and PPAOS had a similar proportion of progressors, although nfvPPA progressed to CBS earlier (p-value = 0.046), driven by an early appearance of limb apraxia (p-value = 0.0041). The non-progressors and progressors both showed premotor/motor cortex involvement at baseline, with spread into prefrontal cortex over time. Discussion: An important proportion of patients with nfvPPA and PPAOS progress to possible CBS, while they rarely develop features of probable CBS even after long follow-up. [ABSTRACT FROM AUTHOR]

Key Clinical Message: Creutzfeldt–Jakob disease is a neurodegenerative disorder caused by brain accumulation of a misfolded form of the cellular prion protein, whose diagnosis is challenging, particularly in early stages, due to the variability and nonspecificity of the clinical and radiological features. 18F‐fluorodeoxyglucose positron‐emitted tomography has the potential to be considered a crucial investigation in these patients, revealing metabolic abnormalities earlier than the conventional neuroimaging analysis. A 59‐year‐old man, the military officer, was referred to our Units for the onset of neurological symptoms rapidly evolving within a month, characterized by akinetic mutism, constructional apraxia, and disorders of spatial orientation. Brain 18F‐fluorodeoxyglucose (18F‐FDG) positron‐emitted tomography (PET)/CT depicted an asymmetric hypometabolism in the left fronto‐temporo‐parietal cortex, as well as in the left thalamus and the right cerebellar hemisphere, while the glucose metabolism appears to be preserved in the somatosensory cortex and the basal ganglia. Laboratory routine analyses, cerebrospinal fluid routine, infective tests, electroencephalography (EEG), and brain magnetic resonance (MR) were all unremarkable. A positive RT‐QuIC result on cerebro‐spinal fluid (CSF) was subsequently shown, without any pathogenic gene mutations and, therefore, the result was consistent with a diagnosis of sporadic Creutzfeld–Jacob disease. The clinical evolution was quickly unfavorable, and the patient died about 4 months after hospital admission. FDG PET/computed tomography (CT) has the potential to be considered a crucial investigation in these patients, documenting metabolic changes long time before other diagnostic investigations such as CSF, EEG, brain CT, and brain MR, thus suggesting a greater sensitivity of glucose metabolic evaluation in the early stage of the disease in question. [ABSTRACT FROM AUTHOR]

Purpose: There are multiple frameworks for goal writing that are applicable to the practice of speech-language pathology. Motor-based speech disorders are a subset of speech sound disorders that are thought to require specific elements of intervention that are typically not addressed in the traditional frameworks used in the clinical setting. The purpose of this tutorial is to review general approaches of goal writing and suggest additional elements that may be used to improve the efficiency and effectiveness of treatment for childhood motor speech disorders, specifically childhood apraxia of speech (CAS). Method: Existing models of goal writing were reviewed to ascertain elements common to most of these models. A basic framework was chosen and modified to include behaviors, conditions, and approaches to goal measurement tailored to the clinical needs of children with CAS. A resource for clinical decision making for children with CAS was developed to inform goal writing at the onset of treatment and adaptations that occur over the course of treatment. Case studies are presented to demonstrate how the presented framework can be applied to writing goals for motor-based treatment for two different children with CAS. Discussion: Children with CAS require a specialized approach to intervention, which requires goals to reflect the unique clinical needs of this population. This tutorial offers resources that use the best available research evidence and current understanding of effective treatment practices for CAS to guide clinical decision making for motor-based intervention and goal writing. This tutorial is intended to guide treatment planning across varied settings to facilitate progress and optimize treatment outcomes for children with CAS. [ABSTRACT FROM AUTHOR]

Background: Perceptual measures such as speech intelligibility are known to be biased, variant and subjective, to which an automatic approach has been seen as a more reliable alternative. On the other hand, automatic approaches tend to lack explainability, an aspect that can prevent the widespread usage of these technologies clinically. Aims: In the present work, we aim to study the relationship between four perceptual parameters and speech intelligibility by automatically modelling the behaviour of six perceptual judges, in the context of head and neck cancer. From this evaluation we want to assess the different levels of relevance of each parameter as well as the different judge profiles that arise, both perceptually and automatically. Methods and Procedures: Based on a passage reading task from the Carcinologic Speech Severity Index (C2SI) corpus, six expert listeners assessed the voice quality, resonance, prosody and phonemic distortions, as well as the speech intelligibility of patients treated for oral or oropharyngeal cancer. A statistical analysis and an ensemble of automatic systems, one per judge, were devised, where speech intelligibility is predicted as a function of the four aforementioned perceptual parameters of voice quality, resonance, prosody and phonemic distortions. Outcomes and Results: The results suggest that we can automatically predict speech intelligibility as a function of the four aforementioned perceptual parameters, achieving a high correlation of 0.775 (Spearman's ρ). Furthermore, different judge profiles were found perceptually that were successfully modelled automatically. Conclusions and Implications: The four investigated perceptual parameters influence the global rating of speech intelligibility, showing that different judge profiles emerge. The proposed automatic approach displayed a more uniform profile across all judges, displaying a more reliable, unbiased and objective prediction. The system also adds an extra layer of interpretability, since speech intelligibility is regressed as a direct function of the individual prediction of the four perceptual parameters, an improvement over more black box approaches. WHAT THIS PAPER ADDS: What is already known on this subject: Speech intelligibility is a clinical measure typically used in the post‐treatment assessment of speech affecting disorders, such as head and neck cancer. Their perceptual assessment is currently the main method of evaluation; however, it is known to be quite subjective since intelligibility can be seen as a combination of other perceptual parameters (voice quality, resonance, etc.). Given this, automatic approaches have been seen as a more viable alternative to the traditionally used perceptual assessments. What this study adds to existing knowledge: The present work introduces a study based on the relationship between four perceptual parameters (voice quality, resonance, prosody and phonemic distortions) and speech intelligibility, by automatically modelling the behaviour of six perceptual judges. The results suggest that different judge profiles arise, both in the perceptual case as well as in the automatic models. These different profiles found showcase the different schools of thought that perceptual judges have, in comparison to the automatic judges, that display more uniform levels of relevance across all the four perceptual parameters. This aspect shows that an automatic approach promotes unbiased, reliable and more objective predictions. What are the clinical implications of this work?: The automatic prediction of speech intelligibility, using a combination of four perceptual parameters, show that these approaches can achieve high correlations with the reference scores while maintaining a certain degree of explainability. The more uniform judge profiles found on the automatic case also display less biased results towards the four perceptual parameters. This aspect facilitates the clinical implementation of this class of systems, as opposed to the more subjective and harder to reproduce perceptual assessments. [ABSTRACT FROM AUTHOR]

GLUT1 deficiency syndrome (GLUT1DS) is a rare genetic disorder caused by a mutation in the SLC2A1 gene that limits the transport of glucose across the blood-brain barrier. Speech disorders and dysarthria are typical findings in patients with GLUT1DS, but have never been deeply phenotyped. The aim of the present study was to characterize speech abilities in a sample of patients with GLUT1DS. 30 patients with GLUT1DS were recruited. We reported impairments in different speech and oromotor domains: the speech was characterized by dysarthria, inaccurate articulation of consonants, abnormal nasal resonance, errors in intonation and prosody and low intelligibility. We observed difficulties in motor planning and programming. Moreover, we observed a significant difference between the dysarthric level of impairment with genotype groups. The presence of a speech disorder in patients with GLUT1DS represents a core feature of the syndrome. Our findings suggest that patients with GLUT1DS would benefit from a comprehensive neurocognitive assessment to detect strengths and weaknesses of the speech profile. Understanding the speech and language phenotype in GLUT1DS is critical for planning early intervention to positively influence the global development of patients with GLUT1DS. • The purpose of this study is to analyze and characterize the speech profile of a sample of patients with GLUT1DS. • The majority of the patients manifested dysarthria with different levels of severity. • A relationship between genotype and dysartric level of impairment was found. • The introduction of the ketogenic diet therapy might be a protective factor for cognitive outcomes in patients with GLUT1DS. [ABSTRACT FROM AUTHOR]

Childhood apraxia of speech (CAS) represents a significant diagnostic and therapeutic challenge within the field of clinical neuropsychology, characterized by its nuanced presentation and multifactorial nature. The aim of this study was to distil and synthesize the broad spectrum of research into a coherent model for the assessment and diagnosis of CAS. Through a mixed-method design, the quantitative phase analyzed 290 studies, unveiling 10 clusters: developmental apraxia, tabby talk, intellectual disabilities, underlying speech processes, breakpoint localization, speech characteristics, functional characteristics, clinical practice, and treatment outcome. The qualitative phase conducted a thematic analysis on the most cited and recent literature, identifying 10 categories: neurobiological markers, speech motor control, perceptual speech features, auditory processing, prosody and stress patterns, parent- and self-report measures, intervention response, motor learning and generalization, comorbidity analysis, and cultural and linguistic considerations. Integrating these findings, a descriptive and prescriptive model was developed, encapsulating the complexities of CAS and providing a structured approach for clinicians. This model advances the understanding of CAS and supports the development of targeted interventions. This study concludes with a call for evidence-based personalized treatment plans that account for the diverse neurobiological and cultural backgrounds of children with CAS. Its implications for practice include the integration of cutting-edge assessment tools that embrace the heterogeneity of CAS presentations, ensuring that interventions are as unique as the children they aim to support. [ABSTRACT FROM AUTHOR]

Progressive apraxia of speech (PAOS) is a 4R tauopathy characterized by difficulties with motor speech planning. Neurodegeneration in PAOS targets the premotor cortex, particularly the supplementary motor area (SMA), with degeneration of white matter (WM) tracts connecting premotor and motor cortices and Broca's area observed on diffusion tensor imaging (DTI). We aimed to assess flortaucipir uptake across speech‐language‐related WM tracts identified using DTI tractography in PAOS. Twenty‐two patients with PAOS and 26 matched healthy controls were recruited by the Neurodegenerative Research Group (NRG) and underwent MRI and flortaucipir‐PET. The patient population included patients with primary progressive apraxia of speech (PPAOS) and non‐fluent variant/agrammatic primary progressive aphasia (agPPA). Flortaucipir PET scans and DTI were coregistered using rigid registration with a mutual information cost function in subject space. Alignments between DTI and flortaucipir PET were inspected in all cases. Whole‐brain tractography was calculated using deterministic algorithms by a tractography reconstruction tool (DSI‐studio) and specific tracts were identified using an automatic fiber tracking atlas‐based method. Fractional anisotropy (FA) and flortaucipir standardized uptake value ratios (SUVRs) were averaged across the frontal aslant tract, arcuate fasciculi, inferior frontal‐occipital fasciculus, inferior and middle longitudinal fasciculi, as well as the SMA commissural fibers. Reduced FA (p <.0001) and elevated flortaucipir SUVR (p =.0012) were observed in PAOS cases compared to controls across all combined WM tracts. For flortaucipir SUVR, the greatest differentiation of PAOS from controls was achieved with the SMA commissural fibers (area under the receiver operator characteristic curve [AUROC] = 0.83), followed by the left arcuate fasciculus (AUROC = 0.75) and left frontal aslant tract (AUROC = 0.71). Our findings demonstrate that flortaucipir uptake is increased across WM tracts related to speech/language difficulties in PAOS. [ABSTRACT FROM AUTHOR]