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Purpose: Spatiotemporal index (STI) is a common measure of articulatory variability used to examine speech-motor control. However, the methods used to elicit productions for measuring STI have varied across studies. The aim of this study was to determine whether STI values are affected by changes in elicitation methods., Method: Lip aperture STI (LA STI) was calculated for 19 monolingual English-speaking young adults based upon the production of four declarative sentences that varied by length and complexity. Using a 2 × 2 design, productions were elicited under the following two conditions: repetition type (consecutive vs. pseudorandom) and stimulus presentation type (auditory vs. combined auditory and visual). Conditions for eliciting productions were counterbalanced among participants., Results: There was a main effect of repetition type ( p < .001) and sentence type ( p < .030). Pseudorandom repetitions resulted in higher mean LA STI values across sentence types compared to those computed from consecutive repetitions. There were no significant differences for stimulus presentation type. That is, no differences in mean LA STI were found between the auditory versus combined auditory and visual presentations., Conclusions: Our findings show that the methods used to elicit sentence productions have a significant effect on LA STI values. Findings suggest that there is a need for researchers to consider these effects when designing methods for measuring LA STI.

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.

Purpose: Pitch variations (tone productions) have been reported as a measure to differentiate Cantonese-speaking children with and without childhood apraxia of speech (CAS). This study aims to examine fundamental frequency ( F 0) changes within syllables and the effects of syllable structure, lexical status, and syllable positions on F 0 in Cantonese-speaking preschool children with and without CAS., Method: Six children with CAS, six children with non-CAS speech sound disorder plus language disorder (S&LD), 22 children with speech sound disorder only (SSD), and 63 children with typical speech-language development (TD) performed the tone sequencing task (TST). Growth curve analysis was employed to analyze and compare the F 0 values within syllables with three Cantonese tones (high level, high rising, and low falling). The analysis considered the effects of syllable structure (vowel and consonant-vowel), lexical status (word and nonword), and syllable position (initial, medial, and final) on F 0, as well as comparisons within and between groups., Results: Within each group, the effects of syllable structure and position on F 0 values were found with different patterns. Between-group comparisons showed that the CAS group had reduced F 0 contrasts. The CAS group could be differentiated from the control groups based on interactions of F 0 with syllable structure and position, but not lexical status. The dissimilarity of F 0 values detected between the CAS and SSD/TD groups was more prominent than that observed between the CAS and S&LD groups., Conclusions: This study demonstrated that Cantonese-speaking children with CAS had difficulty in varying F 0 within syllables as compared to those without CAS, suggesting pitch variation difficulty and language-specific impairment profiles in CAS. Future investigations of objective measures for identifying Cantonese speakers with CAS and cross-linguistic investigations using growth curve analysis and the TST are suggested.

Williams-Beuren syndrome (WBS) and 7q11.23 duplication syndrome (Dup7) are rare neurodevelopmental disorders caused by deletion and duplication of a 1.5 Mb region that includes at least five genes with a known role in epigenetic regulation. We have shown that CNV of this chromosome segment causes dose-dependent, genome-wide changes in DNA methylation, but the specific genes driving these changes are unknown. We measured genome-wide whole blood DNA methylation in six participants with atypical CNV of 7q11.23 (three with deletions and three with duplications) using the Illumina HumanMethylation450k array and compared their profiles with those from groups of individuals with classic WBS or classic Dup7 and with typically developing (TD) controls. Across the top 1000 most variable positions we found that only the atypical rearrangements that changed the copy number of GTF2IRD1 and/or GTF2I (coding for the TFII-IRD1 and TFII-I proteins) clustered with their respective syndromic cohorts. This finding was supported by results from hierarchical clustering across a selection of differentially methylated CpGs, in addition to pyrosequencing validation. These findings suggest that CNV of the GTF2I genes at the telomeric end of the 7q11.23 interval is a key contributor to the large changes in DNA methylation that are seen in blood DNA from our WBS and Dup7 cohorts, compared to TD controls. Our findings suggest that members of the TFII-I protein family are involved in epigenetic processes that alter DNA methylation on a genome-wide level., (© 2023. Springer Nature Limited and Centre of Excellence in Genomic Medicine Research, King Abdulaziz University.)

Limited evidence for early indicators of childhood apraxia of speech (CAS) precludes reliable diagnosis before 36 months, although a few prior studies have identified several potential early indicators. We examined these possible early indicators in 10 toddlers aged 14-24 months at risk for CAS due to a genetic condition: 7q11.23 duplication syndrome (Dup7). Phon Vocalisation analyses were conducted on phonetic transcriptions of each child's vocalisations during an audio-video recorded 30-minute play session with a caregiver and/or a trained research assistant. The resulting data were compared to data previously collected by Overby from similar-aged toddlers developing typically (TD), later diagnosed with CAS (LCAS), or later diagnosed with another speech sound disorder (LSSD). The Dup7 group did not differ significantly from the LCAS group on any measure. In contrast, the Dup7 group evidenced significant delays relative to the LSSD group on canonical babble frequency, volubility, consonant place diversity, and consonant manner diversity and relative to the TD group not only on these measures but also on canonical babble ratio, consonant diversity, and vocalisation structure diversity. Toddlers with Dup7 also demonstrated expressive vocabulary delay as measured by both number of word types orally produced during the play sessions and primary caregivers' responses on a standardised parent-report measure of early expressive vocabulary. Examining babble, phonetic, and phonotactic characteristics from the productions of young children may allow for earlier identification of CAS and a better understanding of the nature of CAS.

Childhood apraxia of speech (CAS) is a paediatric motor speech disorder. We investigated the lexical tone perception and production abilities of children with CAS and the relationships between the two. Three children with CAS, aged between 3;7 and 5;8, were given the Cantonese Tone Identification Test (CANTIT) and the Hong Kong Cantonese Articulation Test (HKCAT) for assessment of tone perception and production, respectively. Accuracy and error patterns were investigated based on their performance on the two tests. Correlation analysis was performed on children's perception and production scores. Two children scored at the lowest rank on the CANTIT, while one child obtained a Z score of 0. All children scored three standard deviations below the mean on the HKCAT. No statistical differences were found among the six tones with respect to perception accuracy, H (5) = 3.731, p = 0.589. Error analysis showed that children with CAS demonstrated more confusion on perceiving tones compared with TD peers. There were no main effects for task ( F (1,2) = 0.040, p = 0.859) or tone (F(5,10 = 0.997, p = 0.467); nor were there task or tone interaction effects on perception versus production accuracy ( F (5,10) = 1.772, p = 0.206). Tone perception and production accuracy were not significantly correlated ( r 2  = 0.181, p = 0.078). Tone perception deficits were evident in two out of three children with CAS, while all children had lexical tone production difficulties. In this small sample, tone production was more universally affected than tone perception.

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., (© 2023. The Author(s).)

Background: A survey study on clinical practice not only provides insight into the implementation of knowledge, but also informs future investigations. There is a limited understanding of childhood apraxia of speech (CAS) in Cantonese speakers. This study examined the clinical practice of CAS in Hong Kong and discussed future directions of research for better evidence-based practice., Methods: Qualified Hong Kong pediatric speech-language pathologists (SLPs) completed the online questionnaire, which had a total of 48 questions regarding their knowledge of and experience with CAS in Cantonese speakers, including assessment, diagnosis, and treatment., Results: Seventy-seven responses were received from Hong Kong SLPs. Most of the SLPs (83.2%) rated their understanding of CAS as either "a little" or "fair". About half (53.2%) of the respondents had worked with children with CAS. No standardized assessment or objective/quantitative measures were used clinically. Instead, seven assessment tasks, including imitation of polysyllabic words and speech and language samples were used commonly. Perceptual judgment of clinical features is still the most popular approach for diagnosis, with a variety of lists in use. Of concern was that, in addition to using some evidence-based approaches, local SLPs treated CAS using approaches that have limited evidence, in the context of less treatment frequency, targeting both speech and language skills within the same session, and with only partial implementation of the approaches., Conclusions: The results suggest that the understanding of CAS among local SLPs requires attention. One reason for this is that evidence regarding the assessment, diagnosis, and treatment of Cantonese speakers with CAS is still limited. Future investigations are needed., Competing Interests: The authors have declared that no competing interests exist., (Copyright: © 2023 Wong et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.)

Purpose: Apraxia of speech (AOS) and childhood apraxia of speech (CAS) are motor-based speech disorders that have been well studied in Indo-European languages. There is limited understanding of these disorders in speakers of Sino-Tibetan languages, such as Chinese. The purpose of this study is to review methods used in research studies for the assessment and diagnosis of AOS and CAS in Chinese speakers., Method: This scoping review followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses Extension for Scoping Reviews guidelines. Articles with a focus on AOS or CAS in Chinese speakers were systematically searched in seven English and six Chinese databases. Three reviewers performed independent screening, data extraction, and quality assessment after obtaining 100% agreement on the prescreening exercise. A qualitative analysis was conducted to rate the quality of diagnoses, ranging from high (Level I) to low (Level III), with Level IV assigned to studies for which the appropriate rating was unclear due to insufficient evidence., Results: Twenty-eight AOS articles and five CAS articles were identified. A variety of assessment and diagnostic methods were reported. No study of Chinese speakers with AOS or CAS received a rating of Level I. The highest level achieved was Level IIIa for both AOS and CAS studies., Conclusions: There is no reliable and valid test or method for the diagnosis of AOS or CAS in Chinese speakers. The current gold standard of diagnosis is based upon expert perceptual judgment. Further single-language and cross-linguistic investigations of AOS and CAS and the future development of assessment and diagnostic methods are recommended.

Background: Childhood apraxia of speech (CAS) is a motor-based speech sound disorder (SSD) with a core impairment in the planning and programming of spatiotemporal parameters of speech movement sequences. CAS may cause deficits in both segmental and suprasegmental components of speech, and it can severely affect children's ability to speak intelligibly and communicate effectively and impact their quality of life. Assessment tasks, such as the maximum performance tasks (MPT) and Syllable Repetition Task (SRT), examine children's segmental sequencing skills to assist with the diagnosis of CAS. In Hong Kong, although the MPT and SRT have been used clinically to diagnose CAS in Cantonese-speaking children, their validity has not been reported. There is an urgent need for such investigations. Suprasegmentally, lexical stress errors have been reported as a consensual feature and to aid in the diagnosis of CAS. However, there are challenges in diagnosing CAS in children who speak tonal languages like Cantonese. A recent study has reported lexical tone errors in Cantonese-speaking children with CAS. Furthermore, deficits in pitch-variation skills were found in Cantonese-speaking children with CAS using a tone sequencing task (TST). It is hypothesized that there is a universal deficit in pitch-variation skills among tonal and nontonal language speakers with CAS. Further investigations of pitch-variation skills using the TST in Cantonese-speaking children with CAS may shed light on suprasegmental deficits in tonal languages and contribute to the development of a valid diagnostic tool for CAS in children who speak other tonal languages, such as Vietnamese, Thai, and Mandarin., Objective: This study aims to examine the diagnostic potential of the MPT, SRT, and TST in diagnosing Cantonese-speaking children with CAS and to investigate pitch-variation skills in Cantonese-speaking children with and without CAS., Methods: A total of 25 children with CAS and 3 groups of age- and gender-matched controls (non-CAS SSD only group, non-CAS SSD co-occurring with language impairment group, and typical development group) will be recruited. All participants will perform the MPT, SRT, and TST measures. Their performances on these tools will be perceptually judged and acoustically measured., Results: Data collection will last from January 1, 2022, to October 30, 2023. As of August 2022, the project has recruited 4 children in the CAS group, 21 children in the non-CAS SSD group, 4 children in the speech and language impairment group, and 53 children in the typical development group., Conclusions: It is anticipated that Cantonese-speaking children with CAS will have poorer pitch-variation skills than the control groups and that the MPT, SRT, and TST will be appropriate diagnostic tools for identifying CAS in Cantonese-speaking children. The project will benefit the field of speech-language pathology locally and internationally, with short- and long-term impacts., International Registered Report Identifier (irrid): DERR1-10.2196/40465., (©Min Ney Wong, Eddy C H Wong, Shelley L Velleman. Originally published in JMIR Research Protocols (https://www.researchprotocols.org), 04.10.2022.)