Capturing cognitive and behavioral variability among individuals with Down syndrome: a latent profile analysis / Marie Moore CHANNELL in Journal of Neurodevelopmental Disorders, 13 (2021)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 13 (2021) Titre : Capturing cognitive and behavioral variability among individuals with Down syndrome: a latent profile analysis Type de document : texte imprimé Auteurs : Marie Moore CHANNELL, Auteur ; Laura J. MATTIE, Auteur ; Debra R. HAMILTON, Auteur ; George T. CAPONE, Auteur ; E. Mark MAHONE, Auteur ; Stephanie L. SHERMAN, Auteur ; Tracie C. ROSSER, Auteur ; Roger H. REEVES, Auteur ; Luther G. KALB, Auteur Langues : Anglais (eng) Mots-clés : Adaptation, Psychological  Adolescent  Adult  Autism Spectrum Disorder  Child  Cognition  Down Syndrome  Executive Function  Female  Humans  Male  Young Adult  Adaptive behavior  Autism symptomatology  Down syndrome  Intellectual disability  Latent profile analysis  Maladaptive behavior  Phenotypes Index. décimale : PER Périodiques Résumé : BACKGROUND: There is a high degree of inter- and intra-individual variability observed within the phenotype of Down syndrome. The Down Syndrome Cognition Project was formed to capture this variability by developing a large nationwide database of cognitive, behavioral, health, and genetic information on individuals with Down syndrome, ages 6-25 years. The current study used the Down Syndrome Cognition Project database to characterize cognitive and behavioral variability among individuals with Down syndrome. METHODS: Latent profile analysis was used to identify classes across a sample of 314 participants based on their cognition (IQ and executive functioning), adaptive and maladaptive behavior, and autism spectrum disorder symptomatology. A multivariate multinomial regression model simultaneously examined demographic correlates of class. RESULTS: Results supported a 3-class model. Each class demonstrated a unique profile across the subdomains of cognition and behavior. The "normative" class was the largest (n = 153, 48%) and displayed a relatively consistent profile of cognition and adaptive behavior, with low rates of maladaptive behavior and autism symptomatology. The "cognitive" class (n = 109, 35%) displayed low cognitive scores and adaptive behavior and more autism symptomatology, but with low rates of maladaptive behavior. The "behavioral" class, the smallest group (n = 52, 17%), demonstrated higher rates of maladaptive behavior and autism symptomatology, but with cognition levels similar to the "normative" class; their adaptive behavior scores fell in between the other two classes. Household income and sex were the only demographic variables to differ among classes. CONCLUSIONS: These findings highlight the importance of subtyping the cognitive and behavioral phenotype among individuals with Down syndrome to identify more homogeneous classes for future intervention and etiologic studies. Results also demonstrate the feasibility of using latent profile analysis to distinguish subtypes in this population. Limitations and future directions are discussed. En ligne : https://dx.doi.org/10.1186/s11689-021-09365-2 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=574 [article] Capturing cognitive and behavioral variability among individuals with Down syndrome: a latent profile analysis [texte imprimé] / Marie Moore CHANNELL, Auteur ; Laura J. MATTIE, Auteur ; Debra R. HAMILTON, Auteur ; George T. CAPONE, Auteur ; E. Mark MAHONE, Auteur ; Stephanie L. SHERMAN, Auteur ; Tracie C. ROSSER, Auteur ; Roger H. REEVES, Auteur ; Luther G. KALB, Auteur. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 13 (2021) Mots-clés : Adaptation, Psychological  Adolescent  Adult  Autism Spectrum Disorder  Child  Cognition  Down Syndrome  Executive Function  Female  Humans  Male  Young Adult  Adaptive behavior  Autism symptomatology  Down syndrome  Intellectual disability  Latent profile analysis  Maladaptive behavior  Phenotypes Index. décimale : PER Périodiques Résumé : BACKGROUND: There is a high degree of inter- and intra-individual variability observed within the phenotype of Down syndrome. The Down Syndrome Cognition Project was formed to capture this variability by developing a large nationwide database of cognitive, behavioral, health, and genetic information on individuals with Down syndrome, ages 6-25 years. The current study used the Down Syndrome Cognition Project database to characterize cognitive and behavioral variability among individuals with Down syndrome. METHODS: Latent profile analysis was used to identify classes across a sample of 314 participants based on their cognition (IQ and executive functioning), adaptive and maladaptive behavior, and autism spectrum disorder symptomatology. A multivariate multinomial regression model simultaneously examined demographic correlates of class. RESULTS: Results supported a 3-class model. Each class demonstrated a unique profile across the subdomains of cognition and behavior. The "normative" class was the largest (n = 153, 48%) and displayed a relatively consistent profile of cognition and adaptive behavior, with low rates of maladaptive behavior and autism symptomatology. The "cognitive" class (n = 109, 35%) displayed low cognitive scores and adaptive behavior and more autism symptomatology, but with low rates of maladaptive behavior. The "behavioral" class, the smallest group (n = 52, 17%), demonstrated higher rates of maladaptive behavior and autism symptomatology, but with cognition levels similar to the "normative" class; their adaptive behavior scores fell in between the other two classes. Household income and sex were the only demographic variables to differ among classes. CONCLUSIONS: These findings highlight the importance of subtyping the cognitive and behavioral phenotype among individuals with Down syndrome to identify more homogeneous classes for future intervention and etiologic studies. Results also demonstrate the feasibility of using latent profile analysis to distinguish subtypes in this population. Limitations and future directions are discussed. En ligne : https://dx.doi.org/10.1186/s11689-021-09365-2 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=574

Early communication development in infants and toddlers with Fragile X syndrome / Laura J. MATTIE in Autism & Developmental Language Impairments, 7 (January-December 2022)  

Public ISBD [article]  inAutism & Developmental Language Impairments > 7 (January-December 2022) Titre : Early communication development in infants and toddlers with Fragile X syndrome Type de document : texte imprimé Auteurs : Laura J. MATTIE, Auteur ; Lisa R. HAMRICK, Auteur Langues : Anglais (eng) Mots-clés : Early communication  fragile X syndrome  language development  early identification  screening measures Index. décimale : PER Périodiques Résumé : Background and AimsIndividuals with fragile X syndrome (FXS) characteristically struggle with language and communication throughout the life course, but there is limited research on the development of communication before 24 months. The purpose of this study is to describe the early communication of infants and toddlers with FXS using the Communication and Symbolic Behavior Scales-Caregiver Questionnaire (CSBS-CQ), a standardized communication screening measure, as compared to the reported normative data of the CSBS-CQ and identify the percentage of infants and toddlers who scored within the range of concern. Documenting how children with FXS perform on screening measures can provide a quick snapshot of skills to help clinicians determine the need for services.MethodsParticipants were 22 infants and toddlers with FXS between 6 and 29 months. Performance on the CSBS-CQ was compared to the measure's normative data. The CSBS-CQ was completed by mothers, and children were administered the Mullen Scales of Early Learning. Because co-occurring autism is common in FXS, the presence of autism was determined using a clinical best estimate procedure.ResultsOverall and within the domains and subdomains of the CSBS-CQ, infants and toddlers with FXS had significantly lower scores than the normative data. Further, 68.2% of our sample was in the range of concern for their overall communication score. The presence of autism led to consistently lower scores, and more infants and toddlers with FXS+autism scored within the range of concern.ConclusionsOur findings suggest that delays in early communication are evident in comparison to typically developing norms before 24 months. These findings also emphasize that infants and toddlers with FXS would likely benefit from early language intervention given that 68.2% of our sample was in the range of concern for their overall communication score.ImplicationsEarly identification and developmental monitoring of children with FXS will help to determine concerns in communication and other domains of development. While early communication broadly may not be an early indicator of autism in FXS, some specific skills, such as eye gaze, may serve as such an indicator. Screening measures, like the CSBS-CQ, may help monitor both early communication impairments and autism symptoms. Infants and toddlers with FXS, regardless of autism status, will benefit from early language interventions. En ligne : https://doi.org/10.1177/23969415221099403 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=477 [article] Early communication development in infants and toddlers with Fragile X syndrome [texte imprimé] / Laura J. MATTIE, Auteur ; Lisa R. HAMRICK, Auteur. Langues : Anglais (eng) in Autism & Developmental Language Impairments > 7 (January-December 2022) Mots-clés : Early communication  fragile X syndrome  language development  early identification  screening measures Index. décimale : PER Périodiques Résumé : Background and AimsIndividuals with fragile X syndrome (FXS) characteristically struggle with language and communication throughout the life course, but there is limited research on the development of communication before 24 months. The purpose of this study is to describe the early communication of infants and toddlers with FXS using the Communication and Symbolic Behavior Scales-Caregiver Questionnaire (CSBS-CQ), a standardized communication screening measure, as compared to the reported normative data of the CSBS-CQ and identify the percentage of infants and toddlers who scored within the range of concern. Documenting how children with FXS perform on screening measures can provide a quick snapshot of skills to help clinicians determine the need for services.MethodsParticipants were 22 infants and toddlers with FXS between 6 and 29 months. Performance on the CSBS-CQ was compared to the measure's normative data. The CSBS-CQ was completed by mothers, and children were administered the Mullen Scales of Early Learning. Because co-occurring autism is common in FXS, the presence of autism was determined using a clinical best estimate procedure.ResultsOverall and within the domains and subdomains of the CSBS-CQ, infants and toddlers with FXS had significantly lower scores than the normative data. Further, 68.2% of our sample was in the range of concern for their overall communication score. The presence of autism led to consistently lower scores, and more infants and toddlers with FXS+autism scored within the range of concern.ConclusionsOur findings suggest that delays in early communication are evident in comparison to typically developing norms before 24 months. These findings also emphasize that infants and toddlers with FXS would likely benefit from early language intervention given that 68.2% of our sample was in the range of concern for their overall communication score.ImplicationsEarly identification and developmental monitoring of children with FXS will help to determine concerns in communication and other domains of development. While early communication broadly may not be an early indicator of autism in FXS, some specific skills, such as eye gaze, may serve as such an indicator. Screening measures, like the CSBS-CQ, may help monitor both early communication impairments and autism symptoms. Infants and toddlers with FXS, regardless of autism status, will benefit from early language interventions. En ligne : https://doi.org/10.1177/23969415221099403 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=477

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