First name
Amanda
Last name
Bennett

Title

Effect of Family Navigation on Diagnostic Ascertainment Among Children at Risk for Autism: A Randomized Clinical Trial From DBPNet.

Year of Publication

2021

Date Published

2021 Jan 11

ISSN Number

2168-6211

Abstract

<p><strong>Importance: </strong>Early identification of autism spectrum disorder (ASD) is associated with improved cognitive and behavioral outcomes. Targeted strategies are needed to support equitable access to diagnostic services to ensure that children from low-income and racial/ethnic minority families receive the benefits of early ASD identification and treatment.</p>

<p><strong>Objective: </strong>To test the efficacy of family navigation (FN), an individually tailored, culturally informed care management strategy, to increase the likelihood of achieving diagnostic ascertainment among young children at risk for ASD.</p>

<p><strong>Design, Setting, and Participants: </strong>This randomized clinical trial of 249 families of children aged 15 to 27 months who had positive screening results for possible ASD was conducted in 11 urban primary care sites in 3 cities. Data collection occurred from February 24, 2015, through November 5, 2018. Statistical analysis was performed on an intent-to-treat basis from November 5, 2018, to July 27, 2020.</p>

<p><strong>Interventions: </strong>Families were randomized to FN or conventional care management (CCM). Families receiving FN were assigned a navigator who conducted community-based outreach to families to address structural barriers to care and support engagement in recommended services. Families receiving CCM were assigned to a care manager, who did limited telephone outreach. Families received FN or CCM after positive initial screening results and for 100 days after diagnostic ascertainment.</p>

<p><strong>Main Outcomes and Measures: </strong>The primary outcome, diagnostic ascertainment, was measured as the number of days from randomization to completion of the child's clinical developmental evaluation, when a diagnosis of ASD or other developmental disorder was determined.</p>

<p><strong>Results: </strong>Among 250 families randomized, 249 were included in the primary analysis (174 boys [69.9%]; mean [SD] age, 22.0 [3.5] months; 205 [82.3%] publicly insured; 233 [93.6%] non-White). Children who received FN had a greater likelihood of reaching diagnostic ascertainment over the course of 1 year (FN, 108 of 126 [85.7%]; CCM, 94 of 123 [76.4%]; unadjusted hazard ratio [HR], 1.39 [95% CI, 1.05-1.84]). Site (Boston, New Haven, and Philadelphia) and ethnicity (Hispanic vs non-Hispanic) moderated the effect of FN (treatment × site interaction; P = .03; Boston: HR, 2.07 [95% CI, 1.31-3.26]; New Haven: HR, 1.91 [95% CI, 0.94-3.89]; and Philadelphia: HR, 0.91 [95% CI, 0.60-1.37]) (treatment × ethnicity interaction; P &lt; .001; Hispanic families: HR, 2.81 [95% CI, 2.23-3.54] vs non-Hispanic families: HR, 1.49 [95% CI, 1.45-1.53]). The magnitude of FN's effect was significantly greater among Hispanic families than among non-Hispanic families (diagnostic ascertainment among Hispanic families: FN, 90.9% [30 of 33], and CCM, 53.3% [16 of 30]; vs non-Hispanic families: FN, 89.7% [35 of 39], and CCM, 77.5% [31 of 40]).</p>

<p><strong>Conclusions and Relevance: </strong>Family navigation improved the likelihood of diagnostic ascertainment among children from racial/ethnic minority, low-income families who were detected as at risk for ASD in primary care. Results suggest differential effects of FN by site and ethnicity.</p>

<p><strong>Trial Registration: </strong>ClinicalTrials.gov Identifier: NCT02359084.</p>

DOI

10.1001/jamapediatrics.2020.5218

Alternate Title

JAMA Pediatr

PMID

33427861

Title

Person Ability Scores as an Alternative to Norm-Referenced Scores as Outcome Measures in Studies of Neurodevelopmental Disorders.

Year of Publication

2020

Number of Pages

475-480

Date Published

2020 11 01

ISSN Number

1944-7558

Abstract

<p>Although norm-referenced scores are essential to the identification of disability, they possess several features which affect their sensitivity to change. Norm-referenced scores often decrease over time among people with neurodevelopmental disorders who exhibit slower-than-average increases in ability. Further, the reliability of norm-referenced scores is lower at the tails of the distribution, resulting in floor effects and increased measurement error for people with neurodevelopmental disorders. In contrast, the person ability scores generated during the process of constructing a standardized test with item response theory are designed to assess change. We illustrate these limitations of norm-referenced scores, and relative advantages of ability scores, using data from studies of autism spectrum disorder and creatine transporter deficiency.</p>

DOI

10.1352/1944-7558-125.6.475

Alternate Title

Am J Intellect Dev Disabil

PMID

33211814

Title

Early Indicators of Creatine Transporter Deficiency.

Year of Publication

2019

Number of Pages

283-285

Date Published

2019 Mar

ISSN Number

1097-6833

Abstract

<p>Early identification is a goal for creatine transporter deficiency and will be critical for future treatment. Before their first birthday, one-half of this sample showed both a significant feeding/weight gain issue and delayed sitting or crawling. Combined, these early indicators could have alerted providers to conduct a urine screen.</p>

DOI

10.1016/j.jpeds.2018.11.008

Alternate Title

J. Pediatr.

PMID

30579583

Title

Accuracy of Autism Screening in a Large Pediatric Network.

Year of Publication

2019

Date Published

2019 Oct

ISSN Number

1098-4275

Abstract

<p><strong>BACKGROUND: </strong>Universal screening is recommended to reduce the age of diagnosis for autism spectrum disorder (ASD). However, there are insufficient data on children who screen negative and no study of outcomes from truly universal screening. With this study, we filled these gaps by examining the accuracy of universal screening with systematic follow-up through 4 to 8 years.</p>

<p><strong>METHODS: </strong>Universal, primary care-based screening was conducted using the Modified Checklist for Autism in Toddlers with Follow-Up (M-CHAT/F) and supported by electronic administration and integration into electronic health records. All children with a well-child visit (1) between 16 and 26 months, (2) at a Children's Hospital of Philadelphia site after universal electronic screening was initiated, and (3) between January 2011 and July 2015 were included ( = 25 999).</p>

<p><strong>RESULTS: </strong>Nearly universal screening was achieved (91%), and ASD prevalence was 2.2%. Overall, the M-CHAT/F's sensitivity was 38.8%, and its positive predictive value (PPV) was 14.6%. Sensitivity was higher in older toddlers and with repeated screenings, whereas PPV was lower in girls. Finally, the M-CHAT/F's specificity and PPV were lower in children of color and those from lower-income households.</p>

<p><strong>CONCLUSIONS: </strong>Universal screening in primary care is possible when supported by electronic administration. In this "real-world" cohort that was systematically followed, the M-CHAT/F was less accurate in detecting ASD than in previous studies. Disparities in screening rates and accuracy were evident in traditionally underrepresented groups. Future research should focus on the development of new methods that detect a greater proportion of children with ASD and reduce disparities in the screening process.</p>

DOI

10.1542/peds.2018-3963

Alternate Title

Pediatrics

PMID

31562252

Title

A mixed-methods process evaluation of Family Navigation implementation for autism spectrum disorder.

Year of Publication

2018

Number of Pages

1362361318808460

Date Published

2018 Nov 08

ISSN Number

1461-7005

Abstract

<p>There is growing interest in Family Navigation as an approach to improving access to care for children with autism spectrum disorder, yet little data exist on the implementation of Family Navigation. The aim of this study was to identify potential failures in implementing Family Navigation for children with autism spectrum disorder, using a failure modes and effects analysis. This mixed-methods study was set within a randomized controlled trial testing the effectiveness of Family Navigation in reducing the time from screening to diagnosis and treatment for autism spectrum disorder across three states. Using standard failure modes and effects analysis methodology, experts in Family Navigation for autism spectrum disorder (n = 9) rated potential failures in implementation on a 10-point scale in three categories: likelihood of the failure occurring, likelihood of not detecting the failure, and severity of failure. Ratings were then used to create a risk priority number for each failure. The failure modes and effects analysis detected five areas for potential "high priority" failures in implementation: (1) setting up community-based services, (2) initial family meeting, (3) training, (4) fidelity monitoring, and (5) attending testing appointments. Reasons for failure included families not receptive, scheduling, and insufficient training time. The process with the highest risk profile was "setting up community-based services." Failure in "attending testing appointment" was rated as the most severe potential failure. A number of potential failures in Family Navigation implementation-along with strategies for mitigation-were identified. These data can guide those working to implement Family Navigation for children with autism spectrum disorder.</p>

DOI

10.1177/1362361318808460

Alternate Title

Autism

PMID

30404548

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