Lingo: an automated, web-based deep phenotyping platform for language ability.

Background: Language and the ability to communicate effectively are key factors in mental health and well-being. Despite this critical importance, research on language is limited by the lack of a scalable phenotyping toolkit. Methods: Here, we describe and showcase Lingo - a flexible online battery of language and nonverbal reasoning skills based on seven widely used tasks (COWAT, picture narration, vocal rhythm entrainment, rapid automatized naming, following directions, sentence repetition, and nonverbal reasoning). The current version of Lingo takes approximately 30 minutes to complete, is entirely open source, and allows for a wide variety of performance metrics to be extracted. We asked > 1,300 individuals from multiple samples to complete Lingo, then investigated the validity and utility of the resulting data. Results: We conducted an exploratory factor analysis across 14 features derived from the seven assessments, identifying five factors. Four of the five factors showed acceptable test-retest reliability (Pearson's R > 0.7). Factor 2 showed the highest reliability (Pearson's R = 0.95) and loaded primarily on sentence repetition task performance. We validated Lingo with objective measures of language ability by comparing performance to gold-standard assessments: CELF-5 and the VABS-3. Factor 2 was significantly associated with the CELF-5 "core language ability" scale (Pearson's R = 0.77, p-value < 0.05) and the VABS-3 "communication" scale (Pearson's R = 0.74, p-value < 0.05). Factor 2 was positively associated with phenotypic and genetic measures of socieconomic status. Interestingly, we found the parents of children with language impairments had lower Factor 2 scores (p-value < 0.01). Finally, we found Lingo factor scores were significantly predictive of numerous psychiatric and neurodevelopmental conditions. Conclusions: Together, these analyses support Lingo as a powerful platform for scalable deep phenotyping of language and other cognitive abilities. Additionally, exploratory analyses provide supporting evidence for the heritability of language ability and the complex relationship between mental health and language.

Language and reading impairments are associated with increased prevalence of non-right-handedness.

Handedness has been studied for association with language-related disorders because of its link with language hemispheric dominance. No clear pattern has emerged, possibly because of small samples, publication bias, and heterogeneous criteria across studies. Non-right-handedness (NRH) frequency was assessed in N = 2503 cases with reading and/or language impairment and N = 4316 sex-matched controls identified from 10 distinct cohorts (age range 6-19 years old; European ethnicity) using a priori set criteria. A meta-analysis (Ncases  = 1994) showed elevated NRH % in individuals with language/reading impairment compared with controls (OR = 1.21, CI = 1.06-1.39, p = .01). The association between reading/language impairments and NRH could result from shared pathways underlying brain lateralization, handedness, and cognitive functions.

Modeling Population and Subject-Specific Growth in a Latent Trait Measured by Multiple Instruments over Time using a Hierarchical Bayesian Framework.

Psychometric growth curve modeling techniques are used to describe a person's latent ability and how that ability changes over time based on a specific measurement instrument. However, the same instrument cannot always be used over a period of time to measure that latent ability. This is often the case when measuring traits longitudinally in children. Reasons may be that over time some measurement tools that were difficult for young children become too easy as they age resulting in floor effects or ceiling effects or both. We propose a Bayesian hierarchical model for such a scenario. Within the Bayesian model we combine information from multiple instruments used at different age ranges and having different scoring schemes to examine growth in latent ability over time. The model includes between-subject variance and within-subject variance and does not require linking item specific difficulty between the measurement tools. The model's utility is demonstrated on a study of language ability in children from ages one to ten who are hard of hearing where measurement tool specific growth and subject-specific growth are shown in addition to a group level latent growth curve comparing the hard of hearing children to children with normal hearing.

The Slow Development of Real-Time Processing: Spoken-Word Recognition as a Crucible for New Thinking About Language Acquisition and Language Disorders.

Words are fundamental to language, linking sound, articulation, and spelling to meaning and syntax; and lexical deficits are core to communicative disorders. Work in language acquisition commonly focuses on how lexical knowledge-knowledge of words' sound patterns and meanings-is acquired. But lexical knowledge is insufficient to account for skilled language use. Sophisticated real-time processes must decode the sound pattern of words and interpret them appropriately. We review work that bridges this gap by using sensitive real-time measures (eye tracking in the visual world paradigm) of school-age children's processing of highly familiar words. This work reveals that the development of word recognition skills can be characterized by changes in the rate at which decisions unfold in the lexical system (the activation rate). Moreover, contrary to the standard view that these real-time skills largely develop during infancy and toddlerhood, they develop slowly, at least through adolescence. In contrast, language disorders can be linked to differences in the ultimate degree to which competing interpretations are suppressed (competition resolution), and these differences can be mechanistically linked to deficits in inhibition. These findings have implications for real-world problems such as reading difficulties and second-language acquisition. They suggest that developing accurate, flexible, and efficient processing is just as important a developmental goal as is acquiring language knowledge.

Language Growth in Children with Mild to Severe Hearing Loss who Received Early Intervention by 3 Months or 6 Months of Age.

Purpose: To evaluate the impact of hearing screening, diagnosis, and early intervention (EI) by 3 months or 6 months of age on language growth trajectories for children with hearing loss (HL) relative to children with normal hearing (NH). Method: We recruited 133 children with mild to severe HL through universal newborn hearing screening records and referrals from audiologists in the United States and 116 children with NH who served as a comparison group. Examiners administered a battery of developmentally appropriate language measures between 12 months and 8 years of age. We constructed latent growth curve models of global language, grammar, and vocabulary using Bayesian statistics. Results: Children with HL demonstrated no significant differences in initial language skills compared to children with NH. Children in the 1-3-6 group also showed no difference in language growth compared to children with NH. The slope for the 1-2-3 group was significantly steeper than children with NH for global language and grammar. Conclusions: This study documents the positive impact of EI on language outcomes in children with congenital HL. It is among the first to provide evidence to support the potential effects of very early intervention by 3 months of age.

A preliminary epidemiologic study of social (pragmatic) communication disorder in the context of developmental language disorder.

BACKGROUND: There is extremely limited population-based research on social (pragmatic) communication disorder (SCD). Population-based samples have the potential to better characterize the SCD phenotype by mitigating confounds and biases that are typical of convenience and clinical samples. AIMS: The aims of this preliminary epidemiologic study were to advance our understanding of the SCD phenotype relative to developmental language disorder (DLD), obtain an estimate of prevalence, identify risk factors and lay the groundwork for future population level research of SCD. METHODS & PROCEDURES: We analysed existing data from the EpiSLI Database to examine social communication skills in 393 8th grade (13-14 years) children with and without a history of DLD. The primary measure used to evaluate SCD was the Children's Communication Checklist (CCC-2). Two case definitions of SCD reflecting DSM-5 criteria were examined. Both definitions involved significant pragmatic impairment, employing a commonly adopted clinical cut-point of 1.5 SD. In one case, pragmatic deficits could occur along with structural language deficits and, in the other case (established using principal component analysis), pragmatic and social skills were disproportionately lower than structural language abilities. OUTCOMES & RESULTS: When using the first case definition, SCD was much more common in children with a history of DLD than without DLD and history of language disorder at kindergarten was a significant risk factor for SCD in adolescence. However, it is important to note that SCD could be found in children with no prior deficits in other aspects of language. When the second definition was employed, SCD was equally distributed across children with and without a history of DLD. Male sex was a significant risk factor using this case definition of SCD. The estimated prevalence of SCD ranged from 7% (SE = 1.5%) to 11% (SE = 1.7%), acknowledging that prevalence depends on the cut-point selected to determine communication disorder. CONCLUSIONS & IMPLICATIONS: These findings contribute to our understanding of the association between SCD and DLD by recognizing varying profiles of pragmatic and social communication difficulties, which in turn may help refine our diagnostic categories. Preliminary prevalence estimates of SCD can serve as an initial guidepost for identification and planning for intervention services for this condition. WHAT THIS PAPER ADDS: What is already known on this subject There is considerable debate about the diagnostic category of SCD and its relation to other neurodevelopmental disorders. What this study adds to existing knowledge Using data from a US-based epidemiologic sample of DLD, this study offers new information about the association between SCD and DLD, provides preliminary estimates of SCD prevalence, and identifies risk factors for SCD. Clinical implications of this study Improved understanding of possible profiles of pragmatic and social communication deficits will help to clarify diagnostic categories and preliminary prevalence estimates may assist with ensuring availability of adequate intervention services.

Procedural and declarative memory brain systems in developmental language disorder (DLD).

The aim of the current study was to examine microstructural differences in white matter relevant to procedural and declarative memory between adolescents/young adults with and without Developmental Language Disorder (DLD) using diffusion tensor imaging (DTI). The findings showed atypical age-related changes in white matter structures in the corticostriatal system, in the corticocerebellar system, and in the medial temporal region in individuals with DLD. Results highlight the importance of considering the age factor in research on DLD. Future studies are needed to examine the developmental relationship between long-term memory and individual differences in language development and learning.

Aided Hearing Moderates the Academic Outcomes of Children With Mild to Severe Hearing Loss.

OBJECTIVES: There are very limited data regarding the spoken language and academic outcomes of children with mild to severe hearing loss (HL) during the elementary school years, and the findings of these studies are inconsistent. None of these studies have examined the possible role of aided hearing in these outcomes. This study used a large cohort of children to examine these outcomes and in particular to examine whether aided hearing moderates the effect of HL with regard to these outcomes. DESIGN: The spoken language, reading, writing, and calculation abilities were measured after second and fourth grades in children with mild to severe HL (children who are hard of hearing; CHH, n = 183) and a group of children with normal hearing (CNH, n = 91) after the completion of second and fourth grades. Also, among the CHH who wore hearing aids, aided better-ear speech intelligibility index values at the age of school entry were obtained. RESULTS: Oral language abilities of the CHH with mild and moderate HL were similar to the CNH at each grade. Children with moderately-severe HL (better-ear pure tone threshold >59 but <76 dB HL) had significantly poorer oral language and reading skills than the CNH at each grade. The children with mild and moderate HL did not differ from the CNH in oral language or reading. No differences were found between the CHH regardless of severity and CNH with regard to spelling, passage writing, or calculation. The degree to which hearing aids provided audible speech information played a moderating role in the oral language outcomes of CHH and this moderation of language mediated the relationship between the unaided hearing ability of the CHH and their academic outcomes. CONCLUSIONS: As a group, children with mild and moderate HL have good outcomes with regard to language and academic performance. Children with moderately-severe losses were less skilled in language and reading than the CNH and CHH children with mild and moderate losses. Audibility provided by hearing aids was found to moderate the effects of HL with respect to these outcomes. These findings emphasize the importance of including the effects of clinical interventions such as aided hearing when examining outcomes of CHH.

Altered brain structures in the dorsal and ventral language pathways in individuals with and without developmental language disorder (DLD).

Developmental Language Disorder (DLD) is a neurodevelopmental disorder characterized by difficulty learning and using language, and this difficulty cannot be attributed to other developmental conditions. The aim of the current study was to examine structural differences in dorsal and ventral language pathways between adolescents and young adults with and without DLD (age range: 14-27 years) using anatomical magnetic resonance imaging (MRI) and diffusion tensor imaging (DTI). Results showed age-related structural brain differences in both dorsal and ventral pathways in individuals with DLD. These findings provide evidence for neuroanatomical correlates of persistent language deficits in adolescents/young adults with DLD, and further suggest that this brain-language relationship in DLD is better characterized by taking account the dynamic course of the disorder along development.

Early Literacy Predictors and Second-Grade Outcomes in Children Who Are Hard of Hearing.

This study contrasted the early literacy outcomes of children who are hard of hearing (CHH) with children with normal hearing (CNH). At age 5, prereading skills of oral language, phonological processing, and print knowledge were examined in CHH (N = 180) and CNH (N = 80). The CHH had poorer oral language and phonological processing abilities than the CNH but comparable knowledge of print. At age 8, measures of word reading, and reading comprehension yielded no differences between CHH (N = 108) and CNH (N = 62) except for reading comprehension for the moderately severe CHH. Reading achievement in CHH was found to exceed predictions based on prereading performance. This resilience was associated with gains in oral language during the early school years.

A real-time mechanism underlying lexical deficits in developmental language disorder: Between-word inhibition.

Eight to 11% of children have a clinical disorder in oral language (Developmental Language Disorder, DLD). Language deficits in DLD can affect all levels of language and persist through adulthood. Word-level processing may be critical as words link phonology, orthography, syntax and semantics. Thus, a lexical deficit could cascade throughout language. Cognitively, word recognition is a competition process: as the input (e.g., lizard) unfolds, multiple candidates (liver, wizard) compete for recognition. Children with DLD do not fully resolve this competition, but it is unclear what cognitive mechanisms underlie this. We examined lexical inhibition-the ability of more active words to suppress competitors-in 79 adolescents with and without DLD. Participants heard words (e.g. net) in which the onset was manipulated to briefly favor a competitor (neck). This was predicted to inhibit the target, slowing recognition. Word recognition was measured using a task in which participants heard the stimulus, and clicked on a picture of the item from an array of competitors, while eye-movements were monitored as a measure of how strongly the participant was committed to that interpretation over time. TD listeners showed evidence of inhibition with greater interference for stimuli that briefly activated a competitor word. DLD listeners did not. This suggests deficits in DLD may stem from a failure to engage lexical inhibition. This in turn could have ripple effects throughout the language system. This supports theoretical approaches to DLD that emphasize lexical-level deficits, and deficits in real-time processing.

Nonverbal Visual Sequential Learning in Children With Cochlear Implants: Preliminary Findings.

The authors tested the hypothesis that children with cochlear implants (CIs) experience domain-general deficits in sequential learning. Twenty children with CIs and 40 children with normal hearing (NH) participated. Participants completed a serial reaction time task that measured implicit sequential learning. During random sequence phases, the CI group had significantly slower reaction times than the NH group. However, there were no significant differences in the rates of sequential learning between groups. Age at implantation was not significantly associated with learning rate in the CI group. Children with CIs demonstrated nonverbal sequential learning that is comparable to children with NH. Contrary to previous research, early auditory deprivation may not be associated with deficits in domain-general sequential learning, but may affect sequential processing. Further investigation is needed to understand the mechanisms underlying the overall delayed reaction times of children with CIs.

Medical Referral Patterns and Etiologies for Children With Mild-to-Severe Hearing Loss.

OBJECTIVES: To (1) identify the etiologies and risk factors of the patient cohort and determine the degree to which they reflected the incidence for children with hearing loss and (2) quantify practice management patterns in three catchment areas of the United States with available centers of excellence in pediatric hearing loss. DESIGN: Medical information for 307 children with bilateral, mild-to-severe hearing loss was examined retrospectively. Children were participants in the Outcomes of Children with Hearing Loss (OCHL) study, a 5-year longitudinal study that recruited subjects at three different sites. Children aged 6 months to 7 years at time of OCHL enrollment were participants in this study. Children with cochlear implants, children with severe or profound hearing loss, and children with significant cognitive or motor delays were excluded from the OCHL study and, by extension, from this analysis. Medical information was gathered using medical records and participant intake forms, the latter reflecting a caregiver's report. A comparison group included 134 children with normal hearing. A Chi-square test on two-way tables was used to assess for differences in referral patterns by site for the children who are hard of hearing (CHH). Linear regression was performed on gestational age and birth weight as continuous variables. Risk factors were assessed using t tests. The alpha value was set at p < 0.05. RESULTS: Neonatal intensive care unit stay, mechanical ventilation, oxygen requirement, aminoglycoside exposure, and family history were correlated with hearing loss. For this study cohort, congenital cytomegalovirus, strep positivity, bacterial meningitis, extracorporeal membrane oxygenation, and loop diuretic exposure were not associated with hearing loss. Less than 50% of children underwent imaging, although 34.2% of those scanned had abnormalities identified. No single imaging modality was preferred. Differences in referral rates were apparent for neurology, radiology, genetics, and ophthalmology. CONCLUSIONS: The OCHL cohort reflects known etiologies of CHH. Despite available guidelines, centers of excellence, and high-yield rates for imaging, the medical workup for children with hearing loss remains inconsistently implemented and widely variable. There remains limited awareness as to what constitutes appropriate medical assessment for CHH.

Alveolar and Postalveolar Voiceless Fricative and Affricate Productions of Spanish-English Bilingual Children With Cochlear Implants.

Purpose: This study investigates the production of voiceless alveolar and postalveolar fricatives and affricates by bilingual and monolingual children with hearing loss who use cochlear implants (CIs) and their peers with normal hearing (NH). Method: Fifty-four children participated in our study, including 12 Spanish-English bilingual CI users (M = 6;0 [years;months]), 12 monolingual English-speaking children with CIs (M = 6;1), 20 bilingual children with NH (M = 6;5), and 10 monolingual English-speaking children with NH (M = 5;10). Picture elicitation targeting /s/, /tʃ/, and /ʃ/ was administered. Repeated-measures analyses of variance comparing group means for frication duration, rise time, and centroid frequency were conducted for the effects of CI use and bilingualism. Results: All groups distinguished the target sounds in the 3 acoustic parameters examined. Regarding frication duration and rise time, the Spanish productions of bilingual children with CIs differed from their bilingual peers with NH. English frication duration patterns for bilingual versus monolingual CI users also differed. Centroid frequency was a stronger place cue for children with NH than for children with CIs. Conclusion: Patterns of fricative and affricate production display effects of bilingualism and diminished signal, yielding unique patterns for bilingual and monolingual CI users.

Neural patterns elicited by sentence processing uniquely characterize typical development, SLI recovery, and SLI persistence.

BACKGROUND: A substantial amount of work has examined language abilities in young children with specific language impairment (SLI); however, our understanding of the developmental trajectory of language impairment is limited. Along with studying the behavioral changes that occur across development, it is important to examine the neural indices of language processing for children with different language trajectories. The current study sought to examine behavioral and neural bases of language processing in adolescents showing three different trajectories: those with normal language development (NL), those exhibiting persistent SLI (SLI-Persistent), and those with a history of SLI who appear to have recovered (SLI-Recovered). METHODS: Through a sentence judgment task, we examined semantic and syntactic processing. Adolescents judged whether or not each sentence was semantically and syntactically correct. Stimuli consisted of naturally spoken sentences that were either correct, contained a semantic verb error, or contained a syntactic verb agreement error. Verb agreement errors consisted of omission and commission violations of the third-person singular -s. Behavioral button-press responses and electroencephalographic recordings were collected. Behavioral judgments and mean amplitude of the N400 and P600 components were examined. RESULTS: Adolescents in the SLI-Persistent group had lower sentence judgment accuracy overall, relative to the NL and SLI-Recovered groups. Accuracy in judging omission and commission syntactic errors were marginally different, with marginally lower accuracy for commission errors. All groups demonstrated an N400 component elicited by semantic violations. However, adolescents in the SLI-Persistent group demonstrated a less robust P600 component for syntactic violations. Furthermore, adolescents in the SLI-Recovered group exhibited a similar neural profile to the NL group for the semantic and syntactic omission violations. However, a unique profile with initial negativity was observed in the SLI-Recovered group in the commission violation condition. CONCLUSIONS: Adolescents with persistent language impairment continue to demonstrate delays in language processing at the behavioral and neural levels. Conversely, the adolescents in the SLI-Recovered group appear to have made gains in language processing skills to overcome their initial impairments. However, our findings suggest that the adolescents in the SLI-Recovered group may have compensatory processing strategies for some aspects of language, as evidenced by a unique event-related potential profile.

Common Genetic Variants in FOXP2 Are Not Associated with Individual Differences in Language Development.

Much of our current knowledge regarding the association of FOXP2 with speech and language development comes from singleton and small family studies where a small number of rare variants have been identified. However, neither genome-wide nor gene-specific studies have provided evidence that common polymorphisms in the gene contribute to individual differences in language development in the general population. One explanation for this inconsistency is that previous studies have been limited to relatively small samples of individuals with low language abilities, using low density gene coverage. The current study examined the association between common variants in FOXP2 and a quantitative measure of language ability in a population-based cohort of European decent (n = 812). No significant associations were found for a panel of 13 SNPs that covered the coding region of FOXP2 and extended into the promoter region. Power analyses indicated we should have been able to detect a QTL variance of 0.02 for an associated allele with MAF of 0.2 or greater with 80% power. This suggests that, if a common variant associated with language ability in this gene does exist, it is likely of small effect. Our findings lead us to conclude that while genetic variants in FOXP2 may be significant for rare forms of language impairment, they do not contribute appreciably to individual variation in the normal range as found in the general population.

Combining growth curves when a longitudinal study switches measurement tools.

When longitudinal studies are performed to investigate the growth of traits in children, the measurement tool being used to quantify the trait may need to change as the subjects' age throughout the study. Changing the measurement tool at some point in the longitudinal study makes the analysis of that growth challenging which, in turn, makes it difficult to determine what other factors influence the growth rate. We developed a Bayesian hierarchical modeling framework that relates the growth curves per individual for each of the different measurement tools and allows for covariates to influence the shapes of the curves by borrowing strength across curves. The method is motivated by and demonstrated by speech perception outcome measurements of children who were implanted with cochlear implants. Researchers are interested in assessing the impact of age at implantation and comparing the growth rates of children who are implanted under the age of two versus those implanted between the ages of two and four.

Examining Procedural Learning and Corticostriatal Pathways for Individual Differences in Language: Testing Endophenotypes of DRD2/ANKK1.

The aim of the study was to explore whether genetic variation in the dopaminergic system is associated with procedural learning and the corticostriatal pathways in individuals with developmental language impairment (DLI). We viewed these two systems as endophenotypes and hypothesized that they would be more sensitive indicators of genetic effects than the language phenotype itself. Thus, we genotyped two SNPs in the DRD2/ANKK1 gene complex, and tested for their associations to the phenotype of DLI and the two endophenotypes. Results showed that individuals with DLI revealed poor procedural learning abilities and abnormal structures of the basal ganglia. Genetic variation in DRD2/ANKK1 was associated with procedural learning abilities and with microstructural differences of the caudate nucleus. The association of the language phenotype with these DRD2/ANKK1 polymorphisms was not significant, but the phenotype was significantly associated with the two endophenotypes. We suggest that procedural learning and the corticostriatal pathways could be used as effective endophenotypes to aid molecular genetic studies searching for genes predisposing to DLI.

The slow developmental time course of real-time spoken word recognition.

This study investigated the developmental time course of spoken word recognition in older children using eye tracking to assess how the real-time processing dynamics of word recognition change over development. We found that 9-year-olds were slower to activate the target words and showed more early competition from competitor words than 16-year-olds; however, both age groups ultimately fixated targets to the same degree. This contrasts with a prior study of adolescents with language impairment (McMurray, Samelson, Lee, & Tomblin, 2010) that showed a different pattern of real-time processes. These findings suggest that the dynamics of word recognition are still developing even at these late ages, and developmental changes may derive from different sources than individual differences in relative language ability.