Dose frequency randomized controlled trial for Dynamic Temporal and Tactile Cueing (DTTC) treatment for childhood apraxia of speech: protocol paper.

BACKGROUND: Childhood apraxia of speech (CAS) is a pediatric motor-based speech sound disorder that requires a specialized approach to intervention. The extant literature on the treatment of CAS commonly recommends intensive treatment using a motor-based approach, with some of the best evidence supporting the use of Dynamic Temporal and Tactile Cueing (DTTC). To date, a rigorous and systematic comparison of high and low dose frequency (i.e., frequency of therapy sessions) has not been undertaken for DTTC, resulting in a lack of evidence to guide decisions about the optimal treatment schedule for this intervention. The current study aims to fill this gap in knowledge by comparing treatment outcomes when dose frequency is varied. METHODS: A randomized controlled trial will be conducted to examine the efficacy of low versus high dose frequency on DTTC treatment outcomes in children with CAS. A target of 60 children, 2;6-7;11 years of age, will be recruited to participate in this study. Treatment will be provided in the community setting by speech-language pathologists who have completed specialized training administering DTTC in a research reliable manner. True randomization with concealed allocation will be used to assign children to either the low or high dose frequency group. Treatment will be administered in 1-h sessions either 4 times per week over a 6-week period (high dose) or 2 times per week over a 12-week period (low dose). To measure treatment gains, probe data will be collected before treatment, during treatment, and 1 day, 1 week, 4 weeks, and 12 weeks post-treatment. Probe data will consist of customized treated words and a standard set of untreated words to assess generalization of treatment gains. The primary outcome variable will be whole word accuracy, encompassing segmental, phonotactic, and suprasegmental accuracy. DISCUSSION: This will be the first randomized controlled trial to evaluate dose frequency for DTTC treatment in children with CAS. TRIAL REGISTRATION: ClinicalTrials.gov identifier NCT05675306, January 6, 2023.

Motor Speech Phenotypes in Children With Epilepsy: Preliminary Findings.

PURPOSE: This exploratory study aimed to characterize motor speech impairments in a small sample of children with epilepsy, both with and without a known seizure etiology. A secondary aim was to evaluate the validity of the Profile for Childhood Apraxia of speech and Dysarthria (ProCAD), a newly developed tool for differential diagnosis of childhood apraxia of speech and dysarthria. METHOD: Thirteen children with seizure disorders completed a comprehensive speech and language assessment. Three expert speech-language pathologists rated the presence of auditory-perceptual features of motor speech impairment using the ProCAD. Motor speech features, diagnoses, and standardized test scores were compared between children with a known seizure etiology and children with idiopathic epilepsy. RESULTS: Nine of the 13 children exhibited motor speech impairment; dysarthria was the most common diagnosis. Most children (11/13) exhibited language impairment. Group comparisons showed that children with a known seizure etiology had more atypical motor speech features and lower language scores than children with idiopathic seizures. CONCLUSION: These preliminary findings suggest a high rate of motor speech impairment among children with epilepsy.

Prologue to the Forum: Care of the Whole Child: Key Considerations When Working With Children With Childhood Apraxia of Speech.

PURPOSE: This prologue introduces the LSHSS Forum: Care of the Whole Child: Key Considerations When Working With Children With Childhood Apraxia of Speech. The goals of the forum are to provide (a) an overview of several co-occurring conditions and challenges that may affect children in this population and (b) methods and materials to enhance diagnostic accuracy and treatment efficacy to help children with childhood apraxia of speech (CAS) to thrive. METHOD: The prologue provides an overview of what it means to care for the whole child and introduces the five articles in the forum, including research and clinical focus articles as well as tutorials. Infographics, assessment templates, video examples, case studies, and treatment goals are included throughout the forum to promote translation from research to practice. CONCLUSION: Children with CAS may experience a breadth of skills, challenges, and diagnoses. By learning (a) to identify possible co-occurring conditions, (b) when to make referrals, and (c) how to best accommodate and treat children when different conditions are present, speech-language pathologists can further increase the quality of care provided and ability to advocate for some of our most vulnerable clients and their families.

An Investigation of Developmental Coordination Disorder Characteristics in Children With Childhood Apraxia of Speech.

PURPOSE: Children with childhood apraxia of speech (CAS) evidence a high rate of co-occurring fine and gross motor deficits. This clinical focus article reports a preliminary investigation of characteristics of developmental coordination disorder (DCD), a neurodevelopmental disorder categorized by poor motor proficiency and functional limitations, in this population. METHOD: Children with CAS underwent a comprehensive motor evaluation using the Movement Assessment Battery for Children-Second Edition, the Developmental Coordination Disorder Questionnaire, and a developmental history questionnaire to determine if they met criteria for a DCD diagnosis as specified in the Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition (DSM-5). RESULTS: Six out of seven participants met DCD criteria based on the DSM-5 criteria. Four of these children had a co-occurring diagnosis of developmental language disorder, and all met criteria for DCD. CONCLUSIONS: Consistent with previous research, the majority of participants demonstrated motor deficits and 85% met criteria for DCD. Despite this high rate of motor deficits, only 57% had previously undergone a physical/occupational therapy evaluation and intervention and only one had a previous diagnosis of DCD. These findings suggest that formal movement assessments are essential for children with a CAS diagnosis. SUPPLEMENTAL MATERIAL: https://doi.org/10.23641/asha.20540193.

A Tool for Differential Diagnosis of Childhood Apraxia of Speech and Dysarthria in Children: A Tutorial.

PURPOSE: While there has been mounting research centered on the diagnosis of childhood apraxia of speech (CAS), little has focused on differentiating CAS from pediatric dysarthria. Because CAS and dysarthria share overlapping speech symptoms and some children have both motor speech disorders, differential diagnosis can be challenging. There is a need for clinical tools that facilitate assessment of both CAS and dysarthria symptoms in children. The goals of this tutorial are to (a) determine confidence levels of clinicians in differentially diagnosing dysarthria and CAS and (b) provide a systematic procedure for differentiating CAS and pediatric dysarthria in children. METHOD: Evidence related to differential diagnosis of CAS and dysarthria is reviewed. Next, a web-based survey of 359 pediatric speech-language pathologists is used to determine clinical confidence levels in diagnosing CAS and dysarthria. Finally, a checklist of pediatric auditory-perceptual motor speech features is presented along with a procedure to identify CAS and dysarthria in children with suspected motor speech impairments. Case studies illustrate application of this protocol, and treatment implications for complex cases are discussed. RESULTS: The majority (60%) of clinician respondents reported low or no confidence in diagnosing dysarthria in children, and 40% reported they tend not to make this diagnosis as a result. Going forward, clinicians can use the feature checklist and protocol in this tutorial to support the differential diagnosis of CAS and dysarthria in clinical practice. CONCLUSIONS: Incorporating this diagnostic protocol into clinical practice should help increase confidence and accuracy in diagnosing motor speech disorders in children. Future research should test the sensitivity and specificity of this protocol in a large sample of children with varying speech sound disorders. Graduate programs and continuing education trainings should provide opportunities to practice rating speech features for children with dysarthria and CAS. SUPPLEMENTAL MATERIAL: https://doi.org/10.23641/asha.19709146.

Retrospective Case-Control Study of Communication and Motor Abilities in 143 Children With Suspected Childhood Apraxia of Speech: Effect of Concomitant Diagnosis.

Purpose: This study sought to determine if children with childhood apraxia of speech (CAS) plus another major diagnosis (CAS+) are equivalent in communication and motor profiles to those with a primary diagnosis of CAS and no indication or report of any other diagnosis (CAS-Primary). Method: This retrospective case-control study included a chart review of 143 children who were suspected of having CAS at Children's Hospital-Wisconsin between 1998 and 2004. Participants were between 30 and 127 months old and included 107 males. Participants were assigned to the suspected CAS-Primary group (n = 114) if they had characteristics of CAS but no other major diagnosis (e.g., galactosemia) and to the CAS+ group (n = 29) if a comorbid diagnosis was present. Groups were compared across demographic, communication, and motor characteristics. Results: Children with CAS+ evidenced more severe motor profiles than those with CAS-Primary, χ2 = (1, n = 122) = 4.952, p = .026, and a small-to-medium effect size (Φ = .201). On average, communication profiles also tended to be more severe among those with CAS+ wherein receptive language was poorer and phonemic inventories were smaller than those with CAS-Primary. Conclusions: These retrospective data suggest that comorbid diagnosis may play an important role in communication and motor development in children with suspected CAS. These exploratory findings should motivate future prospective studies that consider the role of concomitant diagnoses in symptom profile and response to treatment in children with CAS.

Procedural Learning, Grammar, and Motor Skills in Children With Childhood Apraxia of Speech, Speech Sound Disorder, and Typically Developing Speech.

Purpose This case-control study sought to determine if (a) children with childhood apraxia of speech (CAS), other speech sound disorders (SSDs), and typical development would perform differently on a procedural learning assessment and (b) whether grammatical ability would impact group differences. Method Communication, motor, and procedural learning abilities were assessed in 48 children with CAS (n = 13), SSD (n = 20), and typical development (n = 15), between 43 and 97 months of age (M = 66 months, SD = 12 months). Results On average, children with CAS demonstrated grammatical and motor impairments and required an increased number of exposures to the visuospatial sequence to demonstrate procedural learning, compared to peers with SSD or typical development. A subset of children from each group demonstrated an unanticipated procedural learning pattern wherein they evidenced an uptick in reaction time during the second sequenced block. Children with CAS with this pattern still evidenced procedural learning gains by the fifth sequenced block. In contrast, children with SSD and typical development with this pattern showed poor procedural learning outcomes and were characterized by lower scores on language and motor assessments as well. Conclusions This research provides partial support for the procedural learning deficit hypothesis in children with CAS and for a subset of children with SSD as well. Future research should examine the role of a serial reaction time task in identifying children at risk of multisystem communication and motor deficits. Supplemental Material https://doi.org/10.23641/asha.14173532.

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

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

Differential Diagnosis of Apraxia of Speech in Children and Adults: A Scoping Review.

Purpose Despite having distinct etiologies, acquired apraxia of speech (AOS) and childhood apraxia of speech (CAS) share the same central diagnostic challenge (i.e., isolating markers specific to an impairment in speech motor planning/programming). The purpose of this review was to evaluate and compare the state of the evidence on approaches to differential diagnosis for AOS and CAS and to identify gaps in each literature that could provide directions for future research aimed to improve clinical diagnosis of these disorders. Method We conducted a scoping review of literature published between 1997 and 2019, following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses Extension for Scoping Reviews guidelines. For both AOS and CAS, literature was charted and summarized around four main methodological approaches to diagnosis: speech symptoms, quantitative speech measures, impaired linguistic-motor processes, and neuroimaging. Results Results showed that similar methodological approaches have been used to study differential diagnosis of apraxia of speech in adults and children; however, the specific measures that have received the most research attention differ between AOS and CAS. Several promising candidate markers for AOS and CAS have been identified; however, few studies report metrics that can be used to assess their diagnostic accuracy. Conclusions Over the past two decades, there has been a proliferation of research identifying potential diagnostic markers of AOS and CAS. In order to improve clinical diagnosis of AOS and CAS, there is a need for studies testing the diagnostic accuracy of multiple candidate markers, better control over language impairment comorbidity, more inclusion of speech-disordered control groups, and an increased focus on translational work moving toward clinical implementation of promising measures.

Motor Performance in Children With Childhood Apraxia of Speech and Speech Sound Disorders.

Purpose This study sought to determine if (a) children with childhood apraxia of speech (CAS), other speech sound disorders (SSDs), and typical development (TD) would perform differently on a standardized motor assessment and (b) whether comorbid language impairment would impact group differences. Method Speech, language, and motor abilities were assessed in children with CAS (n = 10), SSD (n = 16), and TD (n = 14) between the ages of 43 and 105 months. Motor skills were evaluated using the Movement Assessment Battery for Children-Second Edition (Henderson, Sugden, & Barnett, 2007), a behavioral assessment that is sensitive in identifying fine/gross motor impairments in children with a range of motor and learning abilities. Data were reanalyzed after reclassifying children by language ability. Results The CAS group performed below the normal limit on all components of the motor assessment and more poorly than the TD and SSD groups on Aiming and Catching and Balance. When children were reclassified by language ability, the comorbid CAS + language impairment group performed worse than the SSD-only and TD groups on Manual Dexterity and Balance and worse than the TD group on Aiming and Catching; all 7 children with CAS + language impairment evidenced performance in the disordered range compared to 1 of 3 children in the CAS-only group and 2 of 6 children in the SSD + language impairment group. Conclusions Children with CAS + language impairment appear to be at an increased risk for motor impairments, which may negatively impact social, academic, and vocational outcomes; referrals for motor screenings/assessments should be considered. Findings may suggest a higher order deficit that mediates cognitive-linguistic and motor impairments in this population.

Exploring the Overlap Between Dyslexia and Speech Sound Production Deficits.

Purpose: Children with dyslexia have speech production deficits in a variety of spoken language contexts. In this article, we discuss the nature of speech production errors in children with dyslexia, including those who have a history of speech sound disorder and those who do not, to familiarize speech-language pathologists with speech production-specific risk factors that may help predict or identify dyslexia in young children. Method: In this tutorial, we discuss the role of a phonological deficit in children with dyslexia and how this may manifest as speech production errors, sometimes in conjunction with a speech sound disorder but sometimes not. We also briefly review other factors outside the realm of phonology that may alert the speech-language pathologist to possible dyslexia. Results: Speech-language pathologists possess unique knowledge that directly contributes to the identification and remediation of children with dyslexia. We present several clinical recommendations related to speech production deficits in children with dyslexia. We also review what is known about how and when children with speech sound disorder are most at risk for dyslexia. Conclusion: Speech-language pathologists have a unique opportunity to assist in the identification of young children who are at risk for dyslexia.

Poor Speech Perception Is Not a Core Deficit of Childhood Apraxia of Speech: Preliminary Findings.

Purpose: Childhood apraxia of speech (CAS) is hypothesized to arise from deficits in speech motor planning and programming, but the influence of abnormal speech perception in CAS on these processes is debated. This study examined speech perception abilities among children with CAS with and without language impairment compared to those with language impairment, speech delay, and typically developing peers. Method: Speech perception was measured by discrimination of synthesized speech syllable continua that varied in frequency (/dɑ/-/ɡɑ/). Groups were classified by performance on speech and language assessments and compared on syllable discrimination thresholds. Within-group variability was also evaluated. Results: Children with CAS without language impairment did not significantly differ in syllable discrimination compared to typically developing peers. In contrast, those with CAS and language impairment showed significantly poorer syllable discrimination abilities compared to children with CAS only and typically developing peers. Children with speech delay and language impairment also showed significantly poorer discrimination abilities, with appreciable within-group variability. Conclusions: These findings suggest that speech perception deficits are not a core feature of CAS but rather occur with co-occurring language impairment in a subset of children with CAS. This study establishes the significance of accounting for language ability in children with CAS. Supplemental Materials: https://doi.org/10.23641/asha.5848056.

Speech Inconsistency in Children With Childhood Apraxia of Speech, Language Impairment, and Speech Delay: Depends on the Stimuli.

Purpose: The current research sought to determine (a) if speech inconsistency is a core feature of childhood apraxia of speech (CAS) or if it is driven by comorbid language impairment that affects a large subset of children with CAS and (b) if speech inconsistency is a sensitive and specific diagnostic marker that can differentiate between CAS and speech delay. Method: Participants included 48 children ranging between 4;7 to 17;8 (years;months) with CAS (n = 10), CAS + language impairment (n = 10), speech delay (n = 10), language impairment (n = 9), or typical development (n = 9). Speech inconsistency was assessed at phonemic and token-to-token levels using a variety of stimuli. Results: Children with CAS and CAS + language impairment performed equivalently on all inconsistency assessments. Children with language impairment evidenced high levels of speech inconsistency on the phrase "buy Bobby a puppy." Token-to-token inconsistency of monosyllabic words and the phrase "buy Bobby a puppy" was sensitive and specific in differentiating children with CAS and speech delay, whereas inconsistency calculated on other stimuli (e.g., multisyllabic words) was less efficacious in differentiating between these disorders. Conclusions: Speech inconsistency is a core feature of CAS and is efficacious in differentiating between children with CAS and speech delay; however, sensitivity and specificity are stimuli dependent.

Evidence for the multiple hits genetic theory for inherited language impairment: a case study.

Communication disorders have complex genetic origins, with constellations of relevant gene markers that vary across individuals. Some genetic variants are present in healthy individuals as well as those affected by developmental disorders. Growing evidence suggests that some variants may increase susceptibility to these disorders in the presence of other pathogenic gene mutations. In the current study, we describe eight children with specific language impairment and four of these children had a copy number variant in one of these potential susceptibility regions on chromosome 15. Three of these four children also had variants in other genes previously associated with language impairment. Our data support the theory that 15q11.2 is a susceptibility region for developmental disorders, specifically language impairment.

Reliance on auditory feedback in children with childhood apraxia of speech.

UNLABELLED: Children with childhood apraxia of speech (CAS) have been hypothesized to continuously monitor their speech through auditory feedback to minimize speech errors. We used an auditory masking paradigm to determine the effect of attenuating auditory feedback on speech in 30 children: 9 with CAS, 10 with speech delay, and 11 with typical development. The masking only affected the speech of children with CAS as measured by voice onset time and vowel space area. These findings provide preliminary support for greater reliance on auditory feedback among children with CAS. LEARNING OUTCOMES: Readers of this article should be able to (i) describe the motivation for investigating the role of auditory feedback in children with CAS; (ii) report the effects of feedback attenuation on speech production in children with CAS, speech delay, and typical development, and (iii) understand how the current findings may support a feedforward program deficit in children with CAS.