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1.
J Clin Exp Neuropsychol ; : 1-10, 2024 May 16.
Artículo en Inglés | MEDLINE | ID: mdl-38753819

RESUMEN

INTRODUCTION: Arranging Pictures is a new episodic memory test based on the NIH Toolbox (NIHTB) Picture Sequence Memory measure and optimized for self-administration on a personal smartphone within the Mobile Toolbox (MTB). We describe evidence from three distinct validation studies. METHOD: In Study 1, 92 participants self-administered Arranging Pictures on study-provided smartphones in the lab and were administered external measures of similar and dissimilar constructs by trained examiners to assess validity under controlled circumstances. In Study 2, 1,021 participants completed the external measures in the lab and self-administered Arranging Pictures remotely on their personal smartphones to assess validity in real-world contexts. In Study 3, 141 participants self-administered Arranging Pictures remotely twice with a two-week delay on personal iOS smartphones to assess test-retest reliability and practice effects. RESULTS: Internal consistency was good across samples (ρxx = .80 to .85, p < .001). Test-retest reliability was marginal (ICC = .49, p < .001) and there were significant practice effects after a two-week delay (ΔM = 3.21 (95% CI [2.56, 3.88]). As expected, correlations with convergent measures were significant and moderate to large in magnitude (ρ = .44 to .76, p < .001), while correlations with discriminant measures were small (ρ = .23 to .27, p < .05) or nonsignificant. Scores demonstrated significant negative correlations with age (ρ = -.32 to -.21, p < .001). Mean performance was slightly higher in the iOS compared to the Android group (MiOS = 18.80, NiOS = 635; MAndroid = 17.11, NAndroid = 386; t(757.73) = 4.17, p < .001), but device type did not significantly influence the psychometric properties of the measure. Indicators of potential cheating were mixed; average scores were significantly higher in the remote samples (F(2, 850) = 11.415, p < .001), but there were not significantly more perfect scores. CONCLUSION: The MTB Arranging Pictures measure demonstrated evidence of reliability and validity when self-administered on personal device. Future research should examine the potential for cheating in remote settings and the properties of the measure in clinical samples.

2.
Am J Intellect Dev Disabil ; 129(3): 191-198, 2024 May 01.
Artículo en Inglés | MEDLINE | ID: mdl-38657963

RESUMEN

Measurement invariance (MI) is a psychometric property of an instrument indicating the degree to which scores from an instrument are comparable across groups. In recent years, there has been a marked uptick in publications using MI in intellectual and developmental disability (IDD) samples. Our goal here is to provide an overview of why MI is important to IDD researchers and to describe some challenges to evaluating it, with an eye towards nudging our subfield into a more thoughtful and measured interpretation of studies using MI.


Asunto(s)
Discapacidades del Desarrollo , Discapacidad Intelectual , Psicometría , Humanos , Discapacidad Intelectual/psicología , Psicometría/normas , Psicometría/instrumentación , Investigación Biomédica/normas
3.
Artículo en Inglés | MEDLINE | ID: mdl-38414411

RESUMEN

OBJECTIVE: We describe the development of a new computer adaptive vocabulary test, Mobile Toolbox (MTB) Word Meaning, and validity evidence from 3 studies. METHOD: Word Meaning was designed to be a multiple-choice synonym test optimized for self-administration on a personal smartphone. The items were first calibrated online in a sample of 7,525 participants to create the computer-adaptive test algorithm for the Word Meaning measure within the MTB app. In Study 1, 92 participants self-administered Word Meaning on study-provided smartphones in the lab and were administered external measures by trained examiners. In Study 2, 1,021 participants completed the external measures in the lab and Word Meaning was self-administered remotely on their personal smartphones. In Study 3, 141 participants self-administered Word Meaning remotely twice with a 2-week delay on personal iPhones. RESULTS: The final bank included 1363 items. Internal consistency was adequate to good across samples (ρxx = 0.78 to 0.81, p < .001). Test-retest reliability was good (ICC = 0.65, p < .001), and the mean theta score was not significantly different upon the second administration. Correlations were moderate to large with measures of similar constructs (ρ = 0.67-0.75, p < .001) and non-significant with measures of dissimilar constructs. Scores demonstrated small to moderate correlations with age (ρ = 0.35 to 0.45, p < .001) and education (ρ = 0.26, p < .001). CONCLUSION: The MTB Word Meaning measure demonstrated evidence of reliability and validity in three samples. Further validation studies in clinical samples are necessary.

4.
Res Sq ; 2024 Jan 08.
Artículo en Inglés | MEDLINE | ID: mdl-38260292

RESUMEN

Background: Intellectual and developmental disabilities (IDDs) are associated with both cognitive challenges and difficulties in conceptual, social, and practical areas of living (DSM-5). Individuals with IDD often present with an intellectual disability in addition to a developmental disability such as autism or Down syndrome. Those with IDD may present with deficits in intellectual functioning as well as adaptive functioning that interfere with independence and living skills. The present study sought to examine associations of longitudinal developmental change in domains of cognition (NIH Toolbox Cognition Battery, NIHTB-CB) and adaptive behavior domains (Vineland Adaptive Behavior Scales-3; VABS-3) including Socialization, Communication, and Daily Living Skills (DLS) over a two-year period. Methods: Eligible participants for this multisite longitudinal study included those who were between 6 and 26 years at Visit 1, and who had a diagnosis of, or suspected intellectual disability (ID), including borderline ID. Three groups were recruited, including those with fragile X syndrome, Down syndrome, and other/idiopathic intellectual disability. In order to examine the association of developmental change between cognitive and adaptive behavior domains, bivariate latent change score (BLCS) models were fit to compare change in the three cognitive domains measured by the NIHTB-CB (Fluid, Crystallized, Composite) and the three adaptive behavior domains measured by the VABS-3 (Communication, DLS, and Socialization). Results: Over a two-year period, change in cognition (both Crystalized and Composite) was significantly and positively associated with change in daily living skills. Also, baseline cognition level predicted growth in adaptive behavior, however baseline adaptive behavior did not predict growth in cognition in any model. Conclusions: The present study demonstrated that developmental improvements in cognition and adaptive behavior are associated in children and young adults with IDD, indicating the potential for cross-domain effects of intervention. Notably, improvements in Daily Living Skills on the VABS-3 emerged as a primary area of adaptive behavior that positively related to improvements in cognition. This work provides evidence for the clinical, "real life" meaningfulness of the NIHTB-CB in IDD, and important empirical support for the NIHTB-CB as a fit-for-purpose performance-based outcome measure for this population.

5.
Child Dev ; 2024 Jan 13.
Artículo en Inglés | MEDLINE | ID: mdl-38217474

RESUMEN

Cognitive research with developmental samples requires improved methods that support large-scale, diverse, and open science. This paper offers initial evidence to support the Mobile Toolbox (MTB), a self-administered remote smartphone-based cognitive battery, in youth populations, from a pilot sample of 99 children (Mage = 11.79 years; 36% female; 53% White, 33% Black or African American, 9% Asian, and 15% Hispanic). Completion rates (95%-99%), practice performance (96%-100%), internal consistency (0.60-0.98), and correlations with similar NIHTB measures (0.55-0.77) provide the first evidence to support the MTB in a youth sample, although there were some inconsistencies across measures. Preliminary findings provide promising evidence of the MTB in developmental populations, and further studies are encouraged.

6.
Clin Orthop Relat Res ; 482(2): 244-256, 2024 Feb 01.
Artículo en Inglés | MEDLINE | ID: mdl-37646744

RESUMEN

BACKGROUND: The interpretation of patient-reported outcomes requires appropriate comparison data. Currently, no patient-specific reference data exist for the Patient-Reported Outcome Measurement Information System (PROMIS) Physical Function (PF), Upper Extremity (UE), and Pain Interference (PI) scales for individuals 50 years and older. QUESTIONS/PURPOSES: (1) Can all PROMIS PF, UE, and PI items be used for valid cross-country comparisons in these domains among the United States, the United Kingdom, and Germany? (2) How are age, gender, and country related to PROMIS PF, PROMIS UE, and PROMIS PI scores? (3) What is the relationship of age, gender, and country across individuals with PROMIS PF, PROMIS UE, and PROMIS PI scores ranging from very low to very high? METHODS: We conducted telephone interviews to collect custom PROMIS PF (22 items), UE (eight items), and PI (eight items) short forms, as well as sociodemographic data (age, gender, work status, and education level), with participants randomly selected from the general population older than 50 years in the United States (n = 900), United Kingdom (n = 905), and Germany (n = 921). We focused on these individuals because of their higher prevalence of surgeries and lower physical functioning. Although response rates varied across countries (14% for the United Kingdom, 22% for Germany, and 12% for the United States), we used existing normative data to ensure demographic alignment with the overall populations of these countries. This helped mitigate potential nonresponder bias and enhance the representativeness and validity of our findings. We investigated differential item functioning to determine whether all items can be used for valid crosscultural comparisons. To answer our second research question, we compared age groups, gender, and countries using median regressions. Using imputation of plausible values and quantile regression, we modeled age-, gender-, and country-specific distributions of PROMIS scores to obtain patient-specific reference values and answer our third research question. RESULTS: All items from the PROMIS PF, UE, and PI measures were valid for across-country comparisons. We found clinically meaningful associations of age, gender, and country with PROMIS PF, UE, and PI scores. With age, PROMIS PF scores decreased (age ß Median = -0.35 [95% CI -0.40 to -0.31]), and PROMIS UE scores followed a similar trend (age ß Median = -0.38 [95% CI -0.45 to -0.32]). This means that a 10-year increase in age corresponded to a decline in approximately 3.5 points for the PROMIS PF score-a value that is approximately the minimum clinically important difference (MCID). Concurrently, we observed a modest increase in PROMIS PI scores with age, reaching half the MCID after 20 years. Women in all countries scored higher than men on the PROMIS PI and 1 MCID lower on the PROMIS PF and UE. Additionally, there were higher T-scores for the United States than for the United Kingdom across all domains. The difference in scores ranged from 1.21 points for the PROMIS PF to a more pronounced 3.83 points for the PROMIS UE. Participants from the United States exhibited up to half an MCID lower T-scores than their German counterparts for the PROMIS PF and PROMIS PI. In individuals with high levels of physical function, with each 10-year increase in age, there could be a decrease of up to 4 points in PROMIS PF scores. Across all levels of upper extremity function, women reported lower PROMIS UE scores than men by an average of 5 points. CONCLUSION: Our study provides age-, gender-, and country-specific reference values for PROMIS PF, UE, and PI scores, which can be used by clinicians, researchers, and healthcare policymakers to better interpret patient-reported outcomes and provide more personalized care. These findings are particularly relevant for those collecting patient-reported outcomes in their clinical routine and researchers conducting multinational studies. We provide an internet application ( www.common-metrics.org/PROMIS_PF_and_PI_Reference_scores.php ) for user-friendly accessibility in order to perform age, gender, and country conversions of PROMIS scores. Population reference values can also serve as comparators to data collected with other PROMIS short forms or computerized adaptive tests. LEVEL OF EVIDENCE: Level II, diagnostic study.


Asunto(s)
Medición de Resultados Informados por el Paciente , Extremidad Superior , Femenino , Humanos , Masculino , Extremidad Inferior , Diferencia Mínima Clínicamente Importante , Dolor , Persona de Mediana Edad
7.
J Int Neuropsychol Soc ; 29(10): 933-942, 2023 Dec.
Artículo en Inglés | MEDLINE | ID: mdl-37989561

RESUMEN

OBJECTIVE: Our objective was to evaluate the psychometric properties of the culturally adapted NIH Toolbox African Languages® when used in Swahili and Dholuo-speaking children in western Kenya. METHOD: Swahili-speaking participants were recruited from Eldoret and Dholuo-speaking participants from Ajigo; all were <14 years of age and enrolled in primary school. Participants completed a demographics questionnaire and five fluid cognition tests of the NIH Toolbox® African Languages program, including Flanker, Dimensional Change Card Sort (DCCS), Picture Sequence Memory, Pattern Comparison, and List Sorting tests. Statistical analyses examined aspects of reliability, including internal consistency (in both languages) and test-retest reliability (in Dholuo only). RESULTS: Participants included 479 children (n = 239, Swahili-speaking; n = 240, Dholuo-speaking). Generally, the tests had acceptable psychometric properties for research use within Swahili- and Dholuo-speaking populations (mean age = 10.5; SD = 2.3). Issues related to shape identification and accuracy over speed limited the utility of DCCS for many participants, with approximately 25% of children unable to match based on shape. These cultural differences affected outcomes of reliability testing among the Dholuo-speaking cohort, where accuracy improved across all five tests, including speed. CONCLUSIONS: There is preliminary evidence that the NIH Toolbox ® African Languages potentially offers a valid assessment of development and performance using tests of fluid cognition in Swahili and Dholuo among research settings. With piloting underway across other diverse settings, future research should gather additional evidence on the clinical utility and acceptability of these tests, specifically through the establishment of norming data among Kenyan regions and evaluating these psychometric properties.


Asunto(s)
Cognición , Lenguaje , Humanos , Niño , Adolescente , Kenia , Psicometría , Reproducibilidad de los Resultados , Pruebas Neuropsicológicas
8.
Pediatr Pulmonol ; 58(12): 3416-3427, 2023 Dec.
Artículo en Inglés | MEDLINE | ID: mdl-37701973

RESUMEN

INTRODUCTION: Outpatient monitoring of children using invasive home mechanical ventilation (IHMV) is recommended, but access to care can be difficult. This study tested if remote (home-based) data collection was feasible and acceptable in chronic IHMV management. METHODS: A codesign study was conducted with an IHMV program, home nurses, and English- and Spanish-speaking parent-guardians of children using IHMV (0-17 years; n = 19). After prototyping, parents used a remote patient monitoring (RPM) bundle to collect patient heart rate, respiratory rate (RR), oxygen saturation, end-tidal carbon dioxide (EtCO2 ), and ventilator pressure/volume over 8 weeks. User feedback was analyzed using qualitative methods and the System Usability Scale (SUS). Expected marginal mean differences within patient measures when awake, asleep, or after a break were calculated using mixed effects models. RESULTS: Patients were a median 2.9 years old and 11 (58%) took breaks off the ventilator. RPM data were entered on a mean of 83.7% (SD ± 29.1%) weeks. SUS scores were 84.8 (SD ± 10.5) for nurses and 91.8 (SD ± 10.1) for parents. Over 90% of parents agreed/strongly agreed that RPM data collection was feasible and relevant to their child's care. Within-patient comparisons revealed that EtCO2 (break-vs-asleep 2.55 mmHg, d = 0.79 [0.42-1.15], p < .001; awake-vs-break 1.48, d = -0.49 [0.13-0.84], p = .02) and RR (break-vs-asleep 16.14, d = 2.12 [1.71-2.53], p < .001; awake-vs-break 3.44, d = 0.45 [0.10-0.04], p = .03) were significantly higher during ventilator breaks. CONCLUSIONS: RPM data collection in children with IHMV was feasible, acceptable, and captured clinically meaningful vital sign changes during ventilator breaks, supporting the clinical utility of RPM in IHMV management.


Asunto(s)
Servicios de Atención de Salud a Domicilio , Respiración Artificial , Humanos , Niño , Preescolar , Respiración Artificial/métodos , Dióxido de Carbono , Ventiladores Mecánicos , Monitoreo Fisiológico/métodos
9.
J Speech Lang Hear Res ; 66(6): 2064-2078, 2023 06 20.
Artículo en Inglés | MEDLINE | ID: mdl-37267425

RESUMEN

PURPOSE: Parent instruction in communication facilitation strategies for autistic toddlers relies on assumptions that parents interpret child behaviors in alignment with clinician definitions of communication. The purpose of this study was to identify features of child behaviors that are predictive of alignment in identification of child communication between clinical researchers and mothers of young autistic children. METHOD: Participants were 33 mothers and their autistic children between 18 and 48 months of age. Mothers' and clinical researchers' perceptions of child communication were assessed using a procedure in which mothers and clinical researchers each independently identified child communication in the same ten 1-min video clips of each mother's child. Endorsed communicative acts were coded for the presence of conventional forms (e.g., vocalization) and potentially communicative forms (e.g., body movement). Multilevel binomial regressions, fit with Bayesian inference, were conducted to predict classification of maternal endorsements of child communication based on the presence of conventional and potentially communicative forms as either an aligned act (i.e., act endorsed by mother and clinical researcher as communicative) or a unique maternal endorsement (i.e., act endorsed by mother but not clinical researcher). RESULTS: The presence of vocalization, verbalization, and gesture each significantly predicted increased likelihood of alignment; the presence of eye contact did not. Although repetitive and sensory behaviors significantly increased the likelihood of unique maternal endorsement, affect shifts and body movements each significantly reduced the likelihood of unique maternal endorsement, and hand activity was not significantly predictive of unique maternal endorsement. CONCLUSIONS: Misalignment in mothers' and clinical researchers' identification of communication may be in part due to mothers' endorsement of behavioral forms that are not traditionally classified as part of a child's communication repertoire. Findings emphasize the need to work toward designing communication interventions that consider the ways in which clinicians and parents of autistic children each bring their own interpretive frameworks to the early intervention experience.


Asunto(s)
Trastorno Autístico , Relaciones Madre-Hijo , Femenino , Humanos , Preescolar , Trastorno Autístico/diagnóstico , Teorema de Bayes , Madres , Comunicación , Percepción
10.
Qual Life Res ; 32(10): 2779-2787, 2023 Oct.
Artículo en Inglés | MEDLINE | ID: mdl-37227662

RESUMEN

OBJECTIVE: The objective of this study was to determine the patient-reported outcome measure (PROM) score ranges associated with descriptive labels (i.e., within normal limits, mild, moderate, severe) by using bookmarking methods with orthopedic clinicians and patients who have experienced a bone fracture. STUDY DESIGN AND SETTING: We created vignettes comprised of six items and responses from the Patient-Reported Outcomes Measurement Information System (PROMIS) Upper Extremity Function, Physical Function, and Pain Interference item banks reflecting different levels of severity. Two groups of patients with fractures (n = 11) and two groups of orthopedic clinicians (n = 16) reviewed the vignettes and assigned descriptive labels independently and then discussed as a group until reaching consensus via a videoconference platform. RESULTS: PROMIS Physical Function and Pain Interference thresholds (T = 50, 40, 25/30 and T = 50/55, 60, 65/70, respectively) for patients with bone fractures were consistent with the results from other patient populations. Upper Extremity thresholds were about 10 points (1 SD) more severe (T = 40, 30, 25/20) compared to the other measures. Patient and clinician perspectives were similar. CONCLUSION: Bookmarking methods generated meaningful score thresholds for PROMIS measures. These thresholds between severity categories varied by domain. Threshold values for severity represent important supplemental information to interpret PROMIS scores clinically.


Asunto(s)
Fracturas Óseas , Calidad de Vida , Humanos , Calidad de Vida/psicología , Medición de Resultados Informados por el Paciente , Dolor , Extremidad Superior
11.
Qual Life Res ; 32(8): 2353-2360, 2023 Aug.
Artículo en Inglés | MEDLINE | ID: mdl-36943606

RESUMEN

PURPOSE: The social responsiveness scale (SRS) is frequently used to quantify the autism-related phenotype and is gaining use in health outcomes research. However, it has a high respondent burden (65 items) for large-scale studies. Further, most evaluations of it have focused on the school-age form, not the preschool form. More validity evidence of shortened forms is necessary in the general population to support the broader health outcomes context of use. METHODS: We evaluated the psychometrics of the SRS in 7030 individuals from multiple predominantly neurotypical samples in order to shorten it based on non-autistic sample metrics. Analyses included item factor analysis, differential item functioning (DIF), and multiple-group item response theory (IRT) to place the SRS items on a comparable scale, which was then simulated via computer adaptive testing (CAT) administration. RESULTS: The SRS was broadly unidimensional with few methodological residual dependencies. On average, males had more autistic characteristics than females, and preschoolers had fewer characteristics than school-age children. The final IRT calibration included 45 items equated across forms, and each form had 11 with significant wording discrepancies and 9 items with near-identical wording that exhibited form-related DIF. The CAT simulation suggested a median of 14 items was sufficient to reach a reliable score, demonstrating its feasibility across the range of impairments. CONCLUSION: IRT allows practitioners the ability to get highly reliable scores with fewer items than the full-length SRS. This supports the future application of the SRS in a computer adaptive testing mode in both neurotypical and ASD samples.


Asunto(s)
Computadores , Calidad de Vida , Masculino , Femenino , Humanos , Preescolar , Calidad de Vida/psicología , Evaluación de Resultado en la Atención de Salud , Psicometría/métodos , Simulación por Computador , Encuestas y Cuestionarios , Reproducibilidad de los Resultados
12.
J Neurodev Disord ; 15(1): 4, 2023 01 17.
Artículo en Inglés | MEDLINE | ID: mdl-36650450

RESUMEN

BACKGROUND: For genetic conditions associated with neurodevelopmental disorder (GCAND), developmental domains such as motor ability, thinking and learning, social abilities, and communication are potential intervention targets. Performance on measures of developmental concepts can be expressed using several types of scores. Norm-referenced scores are intended for the diagnostic context, allowing for the identification of impairment relative to age-based expectations, and can exhibit dramatic floor effects when used in individuals with more significant limitations. Person ability scores, which are derived via Rasch analysis or item response theory, are available on many standardized tests and are intended to measure within-person change. However, they have not been used or evaluated as primary endpoints in GCAND clinical trials. In this study, we simulated a series of parallel-arm clinical trials under several chronological age and impairment conditions, to compare empirically the power and type I error rate of operationalizing test performance using ability scores rather than norm-referenced scores. RESULTS: Using the Vineland Adaptive Behavior Scales as the example, we demonstrated an advantage in statistical power of ability scores over norm-referenced scores at extreme levels of impairment. This advantage was at least partially driven by floor effects in norm-referenced scores. For simulated conditions where impairment was less severe, ability scores outperformed norm-referenced scores, but they were more similar. The type I error rate closely approximated the nominal type I error rate of 5% for both scores. CONCLUSION: The results of this simulation demonstrate a substantial power and interpretative advantage of ability scores over norm-referenced scores for studies of GCAND that will enroll participants with high levels of impairment. These results are expected to generalize to studies of developmental concepts, regardless of the etiology or specific test. However, the relative advantage of ability scores is expected to be even greater for tests with a higher floor than the Vineland.


Asunto(s)
Trastornos del Neurodesarrollo , Humanos , Comunicación , Aprendizaje , Trastornos del Neurodesarrollo/diagnóstico , Simulación de Paciente
13.
Am J Speech Lang Pathol ; 32(1): 115-127, 2023 01 11.
Artículo en Inglés | MEDLINE | ID: mdl-36525627

RESUMEN

BACKGROUND: Studies of early caregiver-mediated interventions targeting social communication of young autistic children have yielded variable child outcomes. This study examined the effects of combining two caregiver-mediated interventions on caregiver strategy use and child social communication and language outcomes. METHOD: This was a multisite parallel randomized controlled trial. Participants included 120 caregivers and their autistic children between 24 and 36 months of age. Dyads were randomly assigned to receive a hybrid intervention that combined Enhanced Milieu Teaching (EMT) and Joint Attention, Symbolic Play, Engagement, and Regulation (JASPER) or to a behavior management control condition, each delivered over 6 months. Caregivers in the JASP-EMT group received twice-weekly, in-home, and hour-long sessions. Outcomes were measured at baseline, the end of intervention (T1), and 6 months later (T2) and included a naturalistic language sample procedure, standardized measures, and caregiver report measures. This trial was registered at clinicaltrials.gov (NCT02595697). RESULTS: Child outcomes did not differ between conditions at T1 or T2 for child primary (social communication) or secondary (language, play, and autism symptoms) outcomes. Relative to control group caregivers, intervention group caregivers demonstrated significantly higher use of JASP-EMT strategies at T1 and T2, with the exception of two strategies (Responsiveness and Matched Responsiveness), which were used significantly more by control group caregivers. Neither autism severity nor baseline caregiver responsiveness moderated outcomes. Post hoc analyses revealed significant correlations between specific strategies and all child outcomes. CONCLUSIONS: Twice-weekly caregiver-mediated intervention that taught caregivers of autistic children to use social communication support strategies did not yield significant child outcomes. Future studies should examine possible sources for the lack of main effects including unexpected differences in linguistic features of caregiver input, changes in control group caregiver behavior, and insufficient intervention dosage. SUPPLEMENTAL MATERIAL: https://doi.org/10.23641/asha.21714278.


Asunto(s)
Trastorno Autístico , Humanos , Preescolar , Trastorno Autístico/terapia , Cuidadores , Comunicación , Intervención Educativa Precoz , Lenguaje
14.
Epilepsy Behav ; 137(Pt A): 108953, 2022 12.
Artículo en Inglés | MEDLINE | ID: mdl-36368092

RESUMEN

OBJECTIVE: Developmental and epileptic encephalopathies (DEE) entail moderate to profound impairments in gross motor skills and mobility, which are poorly quantified with clinical outcomes assessments (COA) used in neuro-typical populations. We studied the motor domain of the Adaptive Behavior Assessment System-3 for ages 0-5 years (ABAS) used outside of its intended age range with a focus on raw scores. METHODS: In a cross-sectional survey, 117 parents of children with a variety of DEEs (ages 1-35 years, median = 9) completed the motor domain section of the ABAS. Floor and ceiling effects and associations with epilepsy-related factors were assessed with appropriate parametric and nonparametric statistical techniques. The sensitivity of the ABAS and additional measures of mobility borrowed from the cerebral palsy literature (Functional Activities Questionnaire (FAQ-22) walking level (FAQ-WL)) to different levels of the Functional Mobility Scale was determined. RESULTS: ABAS motor scores corresponded to a median age equivalent of 20.5 months (Inter-Quartile Range (IQR) 8-34). Most raw scores corresponded to standardized scores > 2 standard deviations below the ABAS standardization sample mean. ABAS raw scores demonstrated minimal floor and ceiling effects (<5%). In linear regression models, scores increased with age under 6 years (p < 0.0001) but flattened out thereafter. Scores varied substantially by DEE group (p < 0.001) and decreased with higher convulsive seizure frequency (<0.0001) and number of seizure medications (p < 0.001). ABAS and other motor scores were sensitive to important differences in mobility as represented by the FMS at 5 yards. Further, they correlated with declines in mobility function from 5 to 500 yards. SIGNIFICANCE: An out-of-range COA with raw scores may provide a measure of motor ability and mobility sensitive within the range of moderate to profound impairment seen in patients with DEE. This approach could shorten the time to appropriate COA development and ensure timely clinical trial readiness for novel therapies for rare DEEs.


Asunto(s)
Parálisis Cerebral , Preescolar , Humanos , Lactante , Recién Nacido , Parálisis Cerebral/complicaciones , Estudios Transversales , Destreza Motora , Encuestas y Cuestionarios , Caminata
15.
Appl Dev Sci ; 26(4): 785-798, 2022.
Artículo en Inglés | MEDLINE | ID: mdl-36387581

RESUMEN

The integration of neurodevelopmental perspectives into clinical science has identified irritability as an early dimensional marker of lifespan mental health risk. Elucidating the developmental patterning of irritable behavior is key to differentiating normative variation from risk markers. Accounting for dysregulation and contextual features of irritability is useful for differentiation at preschool age, laying the groundwork for even earlier characterization. We provide initial evidence for the validity of the Multidimensional Assessment Profile of Disruptive Behavior Temper Loss Scale, Infant-Toddler version in two independent samples of 12-18-month-olds from the US. We calibrated the measure using item response theory in a large representative sample, then validated within an independent sample. We characterized the developmental patterning of irritable behaviors and their dimensional spectrum, and demonstrated test-retest reliability, and convergent validity. The MAP-DB-IT is a standardized, dimensional survey assessing irritability that serves as a tool for characterizing the developmental expression of early mental health risk.

16.
Am J Intellect Dev Disabil ; 127(6): 473-484, 2022 11 01.
Artículo en Inglés | MEDLINE | ID: mdl-36306408

RESUMEN

The NIH Toolbox Cognition Battery (NIHTB-CB) was developed for epidemiological and longitudinal studies across a wide age span. Such a tool may be useful for intervention trials in conditions characterized by intellectual disability (ID), such as Williams syndrome (WS). Three NIHTB-CB tasks, including two executive functioning (Flanker, Dimensional Change Card Sort) and one episodic memory (Picture Sequence Memory) task, were given to 47 individuals with WS, ages 4 to 50, to evaluate feasibility (i.e., proportion of valid administrations) in this population. Findings indicated that NIHTB-CB tests showed good feasibility. Flanker and DCCS age-corrected scores were negatively correlated with age and showed floor effects, indicating these scores may not be useful for quantifying performance on these NIHTB-CB tests in ID.


Asunto(s)
Síndrome de Williams , Humanos , Preescolar , Niño , Adolescente , Adulto Joven , Adulto , Persona de Mediana Edad , Estudios de Factibilidad , Pruebas Neuropsicológicas , Cognición , Función Ejecutiva
17.
Am J Speech Lang Pathol ; 31(6): 2759-2769, 2022 11 16.
Artículo en Inglés | MEDLINE | ID: mdl-36306799

RESUMEN

PURPOSE: A multimeasure approach was developed to capitalize on the strengths of two screening measures: the Screening Tool for Autism in Toddlers and Young Children (STAT), an observational measure of social communication, and the Systematic Observation of Red Flags (SORF), a checklist including restricted and repetitive behavior (RRB) items. This approach offers a novel method of identifying autism in toddlers. METHOD: This was a retrospective study of data collected from a multidisciplinary diagnostic program for 24- to 36-month-olds with developmental delays. Raters with autism expertise but naïve to diagnoses applied the SORF to STAT videos. Psychometrics were derived for the SORF on STAT observations and a multiple-measure approach that used a Least Absolute Shrinkage and Selection Operator modeling framework to construct a STAT-SORF RRB Hybrid, retaining SORF RRB items based on individual predictive abilities. RESULTS: The SORF alone correctly classified 84% of the sample (84% sensitivity and 86% specificity). The STAT-SORF RRB Hybrid model, which retained four SORF RRB items, correctly classified 90% of a validation sample (95% sensitivity and 75% specificity). CONCLUSION: These findings highlight the potential utility of using multiple autism identification tools and regression-based scoring to establish presumptive eligibility and facilitate early access to autism interventions.


Asunto(s)
Trastorno del Espectro Autista , Trastorno Autístico , Preescolar , Humanos , Trastorno del Espectro Autista/diagnóstico , Trastorno Autístico/diagnóstico , Tamizaje Masivo/métodos , Psicometría , Estudios Retrospectivos
19.
Am J Speech Lang Pathol ; 31(4): 1755-1770, 2022 07 12.
Artículo en Inglés | MEDLINE | ID: mdl-35749738

RESUMEN

PURPOSE: Family-centered practice (FCP) is a core component of early intervention (EI) associated with improved child and family outcomes, but little is known about community-based speech-language pathologists' (SLPs') inclusion of families in EI. Many caregivers of autistic children experience caregiving-related stress, making these intervention principles especially critical to the provision of optimal services. This study aimed to characterize EI SLPs' use of FCP coaching strategies and the quality of caregiver-SLP relationships. METHOD: Participants included 25 families with an autistic toddler and their EI SLP. One intervention session for each SLP-family dyad was recorded and coded for the SLP's use of FCP coaching strategies. Caregivers and SLPs completed surveys about their working alliance, caregiver perceptions of family-centered care, and SLPs' approach to FCP. RESULTS: SLPs primarily use child-directed strategies without caregiver involvement. When involving caregivers, SLPs infrequently use coaching strategies that are important for caregiver learning and collaboration (e.g., joint planning and guided practice with feedback). However, caregivers perceived their child's services to be highly family-centered, and caregivers and SLPs rated their working alliance to be of high quality. CONCLUSIONS: The presence of strong caregiver-SLP working alliances alongside infrequent usage of effective coaching strategies indicates that SLPs may engage caregivers in ways that are perceived to be highly collaborative but are not optimal for caregiver involvement in all aspects of their child's services (goal setting and implementation of intervention). Consideration of family preferences and SLP beliefs about FCP will inform ways to disseminate FCPs needed to optimize families' capacities to support their child's development. SUPPLEMENTAL MATERIAL: https://doi.org/10.23641/asha.20113550.


Asunto(s)
Trastorno Autístico , Patología del Habla y Lenguaje , Trastorno Autístico/terapia , Cuidadores , Preescolar , Intervención Educativa Precoz , Familia , Humanos , Encuestas y Cuestionarios
20.
Front Public Health ; 10: 827412, 2022.
Artículo en Inglés | MEDLINE | ID: mdl-35493380

RESUMEN

Translation of developmental science discoveries is impeded by numerous barriers at different stages of the research-to-practice pipeline. Actualization of the vast potential of the developmental sciences to improve children's health and development in the real world is imperative but has not yet been fully realized. In this commentary, we argue that an integrated developmental-implementation sciences framework will result in a translational mindset essential for accelerating real world impact. We delineate key principles and methods of implementation science of salience to the developmental science audience, lay out a potential synthesis between implementation and developmental sciences, provide an illustration of the Mental Health, Earlier Partnership (MHE-P), and set actionable steps for realization. Blending these approaches along with wide-spread adoption of the translational mindset has transformative potential for population-level impact of developmental science discovery.


Asunto(s)
Ciencia de la Implementación , Niño , Humanos
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