Speech-Language-Hearing Scholarly Works

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Innovative Family-Based Genetically Informed Series of Analyses of Whole-Exome Data Supports Likely Inheritance for Grammar in Children with Specific Language Impairment
(MDPI, 2023-06-28) Andres, Erin M.; Earnest, Kathleen Kelsey; Xuan, Hao; Zhong, Cuncong; Rice, Mabel L.; Raza, Muhammad Hashim
Individuals with specific language impairment (SLI) struggle with language acquisition despite average non-verbal intelligence and otherwise typical development. One SLI account focuses on grammar acquisition delay. The current study aimed to detect novel rare genetic variants associated with performance on a grammar assessment, the Test of Early Grammatical Impairment (TEGI), in English-speaking children. The TEGI was selected due to its sensitivity and specificity, consistently high heritability estimates, and its absence from all but one molecular genetic study. We performed whole exome sequencing (WES) in eight families with SLI (n = 74 total) and follow-up Sanger sequencing in additional unrelated probands (n = 146). We prioritized rare exonic variants shared by individuals with low TEGI performance (n = 34) from at least two families under two filtering workflows: (1) novel and (2) previously reported candidate genes. Candidate variants were observed on six new genes (PDHA2, PCDHB3, FURIN, NOL6, IQGAP3, and BAHCC1), and two genes previously reported for overall language ability (GLI3 and FLNB). We specifically suggest PCDHB3, a protocadherin gene, and NOL6 are critical for ribosome synthesis, as they are important targets of SLI investigation. The proposed SLI candidate genes associated with TEGI performance emphasize the utility of precise phenotyping and family-based genetic study.
A cross-sectional investigation of disfluencies in typically developing Spanish-English bilingual children
(Elsevier, 2023-06-15) Rojas, Raúl; Irani, Farzan; Gusewski, Svenja; Camacho, Natalia
Purpose This study examined the language skills and the type and frequency of disfluencies in the spoken narrative production of typically developing Spanish-English bilingual children. Method A cross-sectional sample of 106 bilingual children (50 boys; 56 girls) enrolled in kindergarten through Grade 4, produced a total of 212 narrative retell language samples in English and Spanish. A specialized fluency coding system was implemented to index the percentage of total (%TD) and stuttering-like disfluencies (%SLD) in each language. Large-scale reference databases were used to classify children’s dual language proficiency profiles (balanced, English dominant, Spanish dominant) based on language sample analysis measures of morphosyntax and lexical diversity. Results The bilingual Spanish-English children in this study did not demonstrate significant cross-linguistic differences for mean %TD or %SLD. However, the mean %TD and %SLD in both languages exceeded the risk threshold based on monolingual English-speaking norms. English dominant bilingual children demonstrated significantly lower %TD in English than Spanish. Spanish dominant children demonstrated significantly lower %SLD in Spanish than English. Conclusions This study included the largest sample size of bilingual Spanish-English children investigated to date from a fluency perspective. The frequency of disfluencies was found to be variable across participants and change dynamically as a function of grade and dual language proficiency profiles, indicating the need for studies that employ larger sample sizes and longitudinal designs.
Sex differences in infant vocalization and the origin of language
(Cell Press, 2023-06-16) Oller, D. Kimbrough; Gilkerson, Jill; Richards, Jeffrey A.; Hannon, Steve; Griebel, Ulrike; Bowman, Dale D.; Brown, Jane A.; Yoo, Hyunjoo; Warren, Steven F.
Seeking to discern the earliest sex differences in language-related activities, our focus is vocal activity in the first two years of life, following up on recent research that unexpectedly showed boys produced significantly more speech-like vocalizations (protophones) than girls during the first year of life.We now bring a much larger body of data to bear on the comparison of early sex differences in vocalization, data based on automated analysis of all-day recordings of infants in their homes. The new evidence, like that of the prior study, also suggests boys produce more protophones than girls in the first year and offers additional basis for informed speculation about biological reasons for these differences. More broadly, the work offers a basis for informed speculations about foundations of language that we propose to have evolved in our distant hominin ancestors, foundations also required in early vocal development of modern human infants.
Exploring individual differences in musical rhythm and grammar skills in school-aged children with typically developing language
(Nature Research, 2023-02-07) Nitin, Rachana; Gustavson, Daniel E.; Aaron, Allison S.; Boorom, Olivia A.; Bush, Catherine T.; Wiens, Natalie; Vaughan, Chloe; Persici, Valentina; Blain, Scott D.; Soman, Uma; Hambrick, David Z.; Camarata, Stephen M.; McAuley, J. Devin; Gordon, Reyna L.
A growing number of studies have shown a connection between rhythmic processing and language skill. It has been proposed that domain-general rhythm abilities might help children to tap into the rhythm of speech (prosody), cueing them to prosodic markers of grammatical (syntactic) information during language acquisition, thus underlying the observed correlations between rhythm and language. Working memory processes common to task demands for musical rhythm discrimination and spoken language paradigms are another possible source of individual variance observed in musical rhythm and language abilities. To investigate the nature of the relationship between musical rhythm and expressive grammar skills, we adopted an individual differences approach in N = 132 elementary school-aged children ages 5–7, with typical language development, and investigated prosodic perception and working memory skills as possible mediators. Aligning with the literature, musical rhythm was correlated with expressive grammar performance (r = 0.41, p < 0.001). Moreover, musical rhythm predicted mastery of complex syntax items (r = 0.26, p = 0.003), suggesting a privileged role of hierarchical processing shared between musical rhythm processing and children’s acquisition of complex syntactic structures. These relationships between rhythm and grammatical skills were not mediated by prosodic perception, working memory, or non-verbal IQ; instead, we uncovered a robust direct effect of musical rhythm perception on grammatical task performance. Future work should focus on possible biological endophenotypes and genetic influences underlying this relationship.
Editorial Perspective: Maximising the benefits of intervention research for children and young people with developmental language disorder (DLD) – a call for international consensus on standards of reporting in intervention studies for children with and at risk for DLD
(Wiley, 2022-09-20) Frizelle, Pauline; McKean, Cristina; Eadie, Patricia; Ebbels, Susan; Fricke, Silke; Justice, Laura M.; Kunnari, Sari; Leitão, Suze; Morgan, Angela T.; Munro, Natalie; Murphy, Carol‐Anne; Storkel, Holly L.; Van Horne, Amanda Owen
Current methods for reporting interventions do not allow key questions of importance to practitioners, service providers, policy-makers and people with DLD to be answered, and hence limit the implementation of effective interventions in the real world. To extend the existing EQUATOR guidelines to the context of speech language therapy/pathology for children with language disorder and to provide more specific guidance on participants, interventions and outcomes within the CONSORT checklist (used to improve the reporting of randomised controlled trials) and TIDieR (Template for Intervention Description and Replication) to ensure consistency of reporting. We will develop a core team to include representatives from each of the key groups who will either use or be influenced by the final reporting guidance across different countries. To achieve each set of aims, we will conduct reviews of the literature (which present typologies of intervention characteristics in (D)LD and related disorders); carry out focus groups; and use systematic consensus methods such as the Delphi technique, nominal group technique or consensus development conferences. Through the development and adoption of standard intervention reporting criteria, we anticipate that we will overcome the numerous barriers for practitioners, services and policy-makers in applying intervention evidence to practice. We believe that establishing international consensus on reporting guidelines would significantly accelerate progress in DLD research and the ease with which it can be used in clinical practice, by capitalising on the growth in intervention studies to enable international collaboration and new methodologies of data pooling, meta-analyses and cross-study comparisons.
Using Motor Tempi to Understand Rhythm and Grammatical Skills in Developmental Language Disorder and Typical Language Development
(MIT Press, 2023-01-18) Ladányi, Enikő; Novakovic, Michaela; Boorom, Olivia A.; Aaron, Allison S.; Scartozzi, Alyssa C.; Gustavson, Daniel E.; Nitin, Rachana; Bamikole, Peter O.; Vaughan, Chloe; Fromboluti, Elisa Kim; Schuele, C. Melanie; Camarata, Stephen M.; McAuley, J. Devin; Gordon, Reyna L.
Children with developmental language disorder (DLD) show relative weaknesses on rhythm tasks beyond their characteristic linguistic impairments. The current study compares preferred tempo and the width of an entrainment region for 5- to 7-year-old typically developing (TD) children and children with DLD and considers the associations with rhythm aptitude and expressive grammar skills in the two populations. Preferred tempo was measured with a spontaneous motor tempo task (tapping tempo at a comfortable speed), and the width (range) of an entrainment region was measured by the difference between the upper (slow) and lower (fast) limits of tapping a rhythm normalized by an individual’s spontaneous motor tempo. Data from N = 16 children with DLD and N = 114 TD children showed that whereas entrainment-region width did not differ across the two groups, slowest motor tempo, the determinant of the upper (slow) limit of the entrainment region, was at a faster tempo in children with DLD vs. TD. In other words, the DLD group could not pace their slow tapping as slowly as the TD group. Entrainment-region width was positively associated with rhythm aptitude and receptive grammar even after taking into account potential confounding factors, whereas expressive grammar did not show an association with any of the tapping measures. Preferred tempo was not associated with any study variables after including covariates in the analyses. These results motivate future neuroscientific studies of low-frequency neural oscillatory mechanisms as the potential neural correlates of entrainment-region width and their associations with musical rhythm and spoken language processing in children with typical and atypical language development.
Descriptive analysis of seizures and comorbidities associated with fragile X syndrome
(Wiley Open Access, 2022-07-18) Albizua, Igor; Charen, Krista; Shubeck, Lisa; Talboy, Amy; Berry-Kravis, Elizabeth; Kaufmann, Walter E.; Stallworth, Jennifer L.; Drazba, Katy T.; Erickson, Craig A.; Sweeney, John A.; Tartaglia, Nicole; Warren, Steven F.; Hagerman, Randi; Sherman, Stephanie L.; Warren, Stephen T.; Jin, Peng; Allen, Emily G.
Background Fragile X syndrome is characterized by a myriad of physical features, behavioral features, and medical problems. Commonly found behavioral features are hyperactivity, anxiety, socialization difficulties, and ASD. There is also a higher incidence than in the general population of strabismus, otitis media, and mitral valve prolapse. In addition, one of the most common medical problems associated with FXS is an increased risk of seizures. A subset of individuals carrying the full mutation of the FMR1 gene and diagnosed with fragile X syndrome (FXS) are reported to experience seizures, mostly during the first 10 years of their life span. Methods As part of a larger project to identify genetic variants that modify the risk of seizures, we collected clinical information from 49 carriers with FXS who experienced seizures and 46 without seizures. We compared seizure type and comorbid conditions based on the source of data as well as family history of seizures. Results We found that the concordance of seizure types observed by parents and medical specialists varied by type of seizure. The most common comorbid condition among those with seizures was autism spectrum disorder (47% per medical records vs. 33% per parent report compared with 19% among those without seizures per parent report); the frequency of other comorbid conditions did not differ among groups. We found a slightly higher frequency of family members who experienced seizures among the seizure group compared with the nonseizure group. Conclusion This study confirms previously reported features of seizures in FXS, supports additional genetic factors, and highlights the importance of information sources, altogether contributing to a better understanding of seizures in FXS.
Test of Early Grammatical Impairment (TEGI)
(Pearson Publishing, 2001) Rice, Mabel L.; Wexler, Kenneth
The Rice/Wexler Test of Early Grammatical Impairment (Rice/Wexler) is an individually administered clinical tool that can be used for the identification, diagnosis, screening, and follow-up evaluation of grammatical deficits in young children, ages 3 through 8 years, who speak Standard American English (SAE). The test focuses on a particular area of grammar that is known to be difficult for young children with language impairment at the age that this grammatical competence becomes well established in children with normal language acquisition. Poor performance on finiteness marking as measured in the Rice/Wexler can serve as a clinical marker that identifies “affected” children, or children who have impairments in this area of grammar. Early identification of language impairments in young children, especially children without other disabilities (sometimes known as Specific Language Impairment), is critical to ensuring the early intervention required to prepare children for the language demands of the early school years. In combination with assessments of vocabulary and speech development, the Rice/Wexler can provide documentation of fundamental elements of language for children during the preschool years and the school entry and early elementary school years.
Family-Based Whole-Exome Analysis of Specific Language Impairment (SLI) Identifies Rare Variants in BUD13, a Component of the Retention and Splicing (RES) Complex
(MDPI, 2021-12-30) Andres, Erin M.; Earnest, Kathleen Kelsey; Zhong, Cuncong; Rice, Mabel L.; Raza, Muhammad Hashim
Specific language impairment (SLI) is a common neurodevelopmental disorder (NDD) that displays high heritability estimates. Genetic studies have identified several loci, but the molecular basis of SLI remains unclear. With the aim to better understand the genetic architecture of SLI, we performed whole-exome sequencing (WES) in a single family (ID: 489; n = 11). We identified co-segregating rare variants in three new genes: BUD13, APLP2, and NDRG2. To determine the significance of these genes in SLI, we Sanger sequenced all coding regions of each gene in unrelated individuals with SLI (n = 175). We observed 13 additional rare variants in 18 unrelated individuals. Variants in BUD13 reached genome-wide significance (p-value < 0.01) upon comparison with similar variants in the 1000 Genomes Project, providing gene level evidence that BUD13 is involved in SLI. Additionally, five BUD13 variants showed cohesive variant level evidence of likely pathogenicity. Bud13 is a component of the retention and splicing (RES) complex. Additional supportive evidence from studies of an animal model (loss-of-function mutations in BUD13 caused a profound neural phenotype) and individuals with an NDD phenotype (carrying a CNV spanning BUD13), indicates BUD13 could be a target for investigation of the neural basis of language.
A preliminary investigation of parent-reported fiction versus non-fiction book preferences of school-age children with autism spectrum disorder
(SAGE Publications, 2018-10-09) Davidson, Meghan M.; Weismer, Susan Ellis
Background & aims Anecdotal evidence suggests that individuals with autism spectrum disorder prefer non-fiction books over fiction books. The current study was the first to investigate parent-reports of children with autism spectrum disorder’s fiction and non-fiction book preferences and whether these relate to individual differences in social communication, oral language, and/or reading abilities. Method Children (ages 8–14 years, M = 10.89, SD = 1.17) with autism spectrum disorder diagnoses (n = 19) and typically developing peers (n = 21) participated. Children completed standardized measures of social communication, oral language, and reading abilities. Parents reported children’s current favorite book, and from these responses, we coded children’s fiction versus non-fiction book preferences. Main contribution Contrary to anecdotal evidence, children with autism spectrum disorder preferred fiction similar to their typically developing peers. Fiction versus non-fiction book preference was significantly related to social communication abilities across both groups. Children’s oral language and reading abilities were related, as expected, but the evidence for a relationship between social communication and reading comprehension was mixed. Conclusions This study provides preliminary evidence supporting the association of social communication in fiction versus non-fiction book preference, which may be related to children’s comprehension and support the theoretical role of social communication knowledge in narrative/fiction. Implications It should not be assumed that all children with autism spectrum disorder prefer expository/non-fiction or do not read narrative/fiction. Children who prefer non-fiction may need additional social communication knowledge support to improve their understanding of narrative fiction.
Mothers’ perspectives on challenging behaviours in their children with fragile X syndrome
(Taylor & Francis, 2018-09-16) Muller, Kristen; Brady, Nancy C.; Warren, Steven F.; Fleming, Kandace K.
Background: Individuals with intellectual and developmental disabilities are more likely to engage in problem behaviours than peers with typical development. The purpose of this study was to provide descriptive and qualitative information about problem behaviours in children with fragile X syndrome (FXS) and how families respond to these behaviours. Method: We examined interview responses from 53 mothers of 9-year-old children with FXS. Results: Defiance, tantrums, inattention, stereotypy, and aggression were the most frequently reported problem behaviours of children with FXS. Stereotypy, physical aggression, self-injury, and elopement were reported more often by mothers of children with dual diagnoses of FXS and autism than by mothers of children with a single diagnosis of FXS. Conclusions: Results indicated that for many families, by 9 years of age, problem behaviours led to frustration and altered family activities.
Early Predictors of Later Expressive Language in Boys With Fragile X Syndrome
(American Association on Intellectual and Developmental Disabilities, 2018-07-30) Fielding-Gebhardt, Heather; Warren, Steven F.
The predictive ability of early consonant inventory and intentional communication on later expressive language was examined in 36 boys with fragile X syndrome (FXS). Autism symptom severity was included as a potential moderator. Participants were visited in their homes twice over a 6-year period, and mother-child interactions were videotaped, coded, and transcribed behavior by behavior. Consonant inventory and concurrent autism symptom severity were predictive of later number of different words, as was the interaction between the two. Intentional communication was not predictive of number of different words. These findings provide additional specific evidence for differences in foundational language abilities associated with autism symptom severity in boys with FXS. Clinical implications are discussed.
Syntactic and Semantic Specialization and Integration in 5- to 6-Year-Old Children during Auditory Sentence Processing
(MIT Press, 2019-11-29) Wang, Jin; Rice, Mabel L.; Booth, James R.
Previous studies have found specialized syntactic and semantic processes in the adult brain during language comprehension. Young children have sophisticated semantic and syntactic aspects of language, yet many previous fMRI studies failed to detect this specialization, possibly due to experimental design and analytical methods. In this current study, 5- to 6-year-old children completed a syntactic task and a semantic task to dissociate these two processes. Multivoxel pattern analysis was used to examine the correlation of patterns within a task (between runs) or across tasks. We found that the left middle temporal gyrus showed more similar patterns within the semantic task compared with across tasks, whereas there was no difference in the correlation within the syntactic task compared with across tasks, suggesting its specialization in semantic processing. Moreover, the left superior temporal gyrus showed more similar patterns within both the semantic task and the syntactic task as compared with across tasks, suggesting its role in integration of semantic and syntactic information. In contrast to the temporal lobe, we did not find specialization or integration effects in either the opercular or triangular part of the inferior frontal gyrus. Overall, our study showed that 5- to 6-year-old children have already developed specialization and integration in the temporal lobe, but not in the frontal lobe, consistent with developmental neurocognitive models of language comprehension in typically developing young children.
Communication in Young Children with Fragile X Syndrome: A Qualitative Study of Mothers’ Perspectives
(American Speech-Language-Hearing Association, 2006-11-01) Brady, Nancy C.; Skinner, Debra; Roberts, Joanne; Hennon, Elizabeth
Purpose: The purposes of the study were to provide descriptive and qualitative information about communication in young children with fragile X syndrome (FXS) and about how families react to and accommodate communication differences in their children. Method: In-depth interviews were conducted with 55 mothers of young children with FXS. Interviewers asked mothers to describe their children’s communication, strategies they used to help promote their children’s communication, communication-related frustrations, their expectations for their children and the roles that they perceive for themselves. Results: Over half the children were nonverbal and learning to communicate with augmentative or alternative communication (AAC). Mothers reported using strategies that were developmentally appropriate and recommended by early childhood experts, such as reading and talking to their children. Many mothers identified challenges faced in helping their child to communicate, and some cited difficulty obtaining speech-language services as a challenge. Mothers identified their roles as caregiver, teacher, therapist and advocate. Conclusions: The perspectives offered by mothers are valuable because they indicate how children with FXS communicate in natural contexts. Information about mothers’ expectations and roles may help clinicians to be sensitive to variables that will impact working with young children and their families.
Task and Participant Variables Predict Communication Complexity Scores (CCS): Closer Examination of the CCS
(American Association on Intellectual and Developmental Disabilities, 2019-11-01) Fleming, Kandace K.; Brady, Nancy C.
Communication Complexity Scale (CCS) scores for 269 minimally verbal participants were examined to determine if communicator behavior and task and communicator characteristics were related to scores in a manner consistent with theoretical and research evidence expectations. Each participant completed an interactive assessment with 6 joint attention tasks and 6 behavior regulation tasks. Caregivers completed the Vineland Adaptive Behavior Scales. Results indicated (a) joint attention tasks yielded lower scores than behavior regulation tasks, (b) older participants had lower scores, (c) individuals with autism spectrum disorder scored more similarly than those without, (d) the difference between joint attention and behavior regulation scores was greater for the autism spectrum disorder group, and (e) adaptive behavior was significantly positively related to complexity scores.
Developmental trajectory of communication repair in children with Fragile X Syndrome
(SAGE Publications, 2020-02-27) Fielding-Gebhardt, Heather; Warren, Steven F.; Brady, Nancy C.
Background and aims The development of communicative competence requires both language and social skills. The ability to repair following a communication breakdown is critical for continued conversational interchange and to ensure comprehension of bids for communication. Communication repair demonstrates adequate language and social skills. Children with Fragile X Syndrome have difficulty with language development and social skills, which may result in delays or deficits in repair. Repair may be additionally impaired in children with Fragile X Syndrome and co-morbid autism. This study examined the development of repair in children with Fragile X Syndrome from toddlerhood into middle childhood. Methods Fifty-five children with Fragile X Syndrome and their biological mothers participated. Data were collected during in-home visits approximately every 18 months. Videotaped mother–child interactions were collected, as well as standardized assessments of language, social skills, and autism symptomology. Results Children with Fragile X Syndrome acquired the ability to repair at 90% mastery by three-and-a-half years of age. Multilevel logistic regressions predicting probability of repair indicated marginally significant effects of mean length of utterance and number of different words, and significant effects of global social skills and autism symptomology. Effect sizes were small to moderate. Conclusions Ability to repair was measured in a naturalistic setting, which allowed children with Fragile X Syndrome to utilize repairs in their daily interactions. Although children with Fragile X Syndrome may have delayed development of repair relative to typically developing expectations, in general they nonetheless catch up and demonstrate a robust ability to repair by three-and-a-half years of age. However, this study provides evidence that individual differences in language and social skills may influence ability to repair in children with Fragile X Syndrome. Finally, the relationship between autism symptoms and repair remains unclear, necessitating further exploration. Implications: Given the noted delay in repair in young children with Fragile X Syndrome, clinicians working with this population should target development of this skill as early as possible to maximize successful social interactions. This may be particularly necessary for children with Fragile X Syndrome and co-morbid autism.
The Impact of Dose and Dose Frequency on Word Learning by Kindergarten Children With Developmental Language Disorder During Interactive Book Reading
(American Speech-Language-Hearing Association, 2019-10-10) Storkel, Holly L.; Komesidou, Rouzana; Pezold, Mollee J.; Pitt, Adrienne R.; Fleming, Kandace K.; Romine, Rebecca Swinburne
Purpose The goal was to determine whether interactive book reading outcomes for children with developmental language disorder (DLD) were affected by manipulation of dose (i.e., the number of exposures to the target word during a book reading session) and dose frequency (i.e., the number of repeated book reading sessions) and whether pretreatment factors predicted treatment response variation. Method Thirty-four kindergarten children with DLD (aged 5;0–6;2 [years;months]) were taught 1 set of words using the Dose 6 and Dose Frequency 6 format from a prior study (Storkel, Voelmle, et al., 2017) and taught a different set of words using an alternative format, either Dose 4 × Dose Frequency 9 or Dose 9 × Dose Frequency 4, determined through random assignment. Word learning was tracked for each treatment via a definition task prior to, during, and after treatment. Results Results showed that children with DLD learned a significant number of words during treatment regardless of the dose and dose frequency format but that significant forgetting of newly learned words occurred in all formats once treatment was withdrawn. Individual differences in word learning were related to Clinical Evaluation of Language Fundamentals Core Language and Understanding Spoken Paragraphs scores. Conclusion When administered at an adequate intensity, variation in the dose and dose frequency of interactive book reading does not appear to influence word learning by children with DLD. Although interactive book reading continues to show promise as an effective word learning intervention for children with DLD, further development is needed to enhance the effectiveness of this treatment approach.
Assessing Oromotor Capacity in ALS: The Effect of a Fixed-Target Task on Lip Biomechanics
(Frontiers Media, 2019-12-05) Eshghi, Marziye; Stipancic, Kaila L.; Mefferd, Antje; Rong, Panying; Berry, James D.; Yunusova, Yana; Green, Jordan R.
Alternating motion rate (AMR) is a standard measure often included in neurological examinations to assess orofacial neuromuscular integrity. AMR is typically derived from recordings of patients producing repetitions of a single syllable as fast and clear as possible on one breath. Because the task places high demands on oromotor performance, particularly articulatory speed, AMRs are widely considered to be tests of maximum performance and, therefore, likely to reveal underlying neurologic deficits. Despite decades of widespread use, biomechanical studies have shown that speakers often circumvent the presumed speed challenge of the standard AMR task. Specifically, speakers are likely to manipulate their displacements (movement amplitude) instead of speed because this strategy requires less motor effort. The current study examined the effectiveness of a novel fixed-target paradigm for minimizing the truncation of articulatory excursions and maximizing motor effort. We compared the standard AMR task to that of a fixed-target AMR task and focused specifically on the tasks' potential to detect decrements in lip motor performance in persons with dysarthria due to amyotrophic lateral sclerosis (ALS). Our participants were 14 healthy controls and 17 individuals with ALS. For the standard AMR task, participants were instructed to produce the syllable /bα/ as quickly and accurately as possible on one breath. For the fixed-target AMR task, participants were given the same instructions, but were also required to strike a physical target placed under the jaw during the opening phase of each syllable. Lip kinematic data were obtained using 3D electromagnetic articulography. 16 kinematic features were extracted using an algorithmic approach. Findings revealed that compared to the standard task, the fixed-target AMR task placed increased motor demands on the oromotor system by eliciting larger excursions, faster speeds, and greater spatiotemporal variability. In addition, participants with ALS exhibited limited ability to adapt to the higher articulatory demands of the fixed-target task. Between the two AMR tasks, the maximum speed during the fixed-target task showed a moderate association with the ALSFRS-R bulbar subscore. Employment of both standard and fixed-target AMR tasks is, however, needed for comprehensive assessment of oromotor function and for elucidating profiles of task adaptation.
Prenatal and perinatal risks for late language emergence in a population-level sample of twins at age 2
(BMC, 2018-02-07) Taylor, Catherine L.; Rice, Mabel L.; Christensen, Daniel; Blair, Eve; Zubrick, Stephen R.
Background Late Language Emergence (LLE) in the first two years of life is one of the most common parental concerns about child development and reasons for seeking advice from health professionals. LLE is much more prevalent in twins (38%) than singletons (20%). In studies of language development in twins without overt disability, adverse prenatal and perinatal environments have been reported to play a lesser role in the etiology of LLE than adverse postnatal environments. However, there is a lack of population-level evidence about prenatal and perinatal risk factors for LLE in twins. This study investigated the extent to which prenatal and perinatal risk factors were associated with LLE in a population-level sample of twins at age 2 without overt disability. Methods The sample comprised 473 twin pairs drawn from a population sample frame comprising statutory notifications of all births in Western Australia (WA), 2000–2003. Twin pairs in which either twin had a known developmental disorder or exposure to language(s) other than English were excluded. Of the 946 twins, 47.9% were male. There were 313 dizygotic and 160 monozygotic twin pairs. LLE was defined as a score at or below the gender-specific 10th percentile on the MacArthur Communicative Development Inventories: Words and Sentences (CDI-WS) (Words Produced). Bivariate and multivariable logistic regression was used to investigate risk factors associated with LLE. Results In the multivariable model, risk factors for LLE in order of decreasing magnitude were: Gestational diabetes had an adjusted odds ratio (aOR) of 19.5 (95% confidence interval (CI) 1.2, 313.1); prolonged TSR (aOR: 13.6 [2.0, 91.1]); multiparity (aOR: 7.6 [1.6, 37.5]), monozygosity (aOR: 6.9 [1.7, 27.9]) and fetal growth restriction (aOR: 4.6 [1.7, 12.7]). Sociodemographic risk factors (e.g., low maternal education, socioeconomic area disadvantage) were not associated with increased odds of LLE. Conclusions The results suggest that adverse prenatal and perinatal environments are important in the etiology of LLE in twins at age 2. It is important that health professionals discuss twin pregnancy and birth risks for delayed speech and language milestones with parents and provide ongoing developmental monitoring for all twins, not just twins with overt disability.
The Unlock Project: A Python-based framework for practical brain-computer interface communication “app” development
(Institute of Electrical and Electronics Engineers, 2012-08) Brumberg, Jonathan S.; Lorenz, Sean D.; Galbraith, Byron V.; Guenther, Frank H.
In this paper we present a framework for reducing the development time needed for creating applications for use in non-invasive brain-computer interfaces (BCI). Our framework is primarily focused on facilitating rapid software “app” development akin to current efforts in consumer portable computing (e.g. smart phones and tablets). This is accomplished by handling intermodule communication without direct user or developer implementation, instead relying on a core subsystem for communication of standard, internal data formats. We also provide a library of hardware interfaces for common mobile EEG platforms for immediate use in BCI applications. A use-case example is described in which a user with amyotrophic lateral sclerosis participated in an electroencephalography-based BCI protocol developed using the proposed framework. We show that our software environment is capable of running in real-time with updates occurring 50–60 times per second with limited computational overhead (5 ms system lag) while providing accurate data acquisition and signal analysis.

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