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Autism Res. 2017 Mar;10(3):485-501. doi: 10.1002/aur.1659. Epub 2016 Aug 3.

Development and validation of a streamlined autism case confirmation approach for use in epidemiologic risk factor research in prospective cohorts.

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A.J. Drexel Autism Institute, Drexel University, 3020 Market Street Suite 560, Philadelphia, PA, 19104.
Center on Human Development and Disability, University of Washington, Box 357920, Seattle, WA, 98195-7920.
Center for Autism and Related Disorders, Kennedy Krieger Institute, 3901 Greenspring Avenue, Baltimore, MD, 21211.
University of California San Francisco School of Medicine, 401 Parnassus Avenue, San Francisco, CA, 94143.
Battelle Centers for Public Health Research and Evaluation, 6115 Falls Road, Baltimore, MD, 21209.
Ichan School of Medicine at Mount Sinai, 17 East 102 Street Floor 2nd Floor - West Tower Room D2-127, New York, NY, 10029.
University of California Los Angeles School of Medicine, UCLA Center for Health of Children, Families and Communities, 1100 Glendon Avenue, Suite 850, Los Angeles, CA, 90024.
University of California Irvine School of Medicine, 252 Irvine Hall, Irvine, CA, 92697.
University of Miami, P.O. Box 249229, Coral Gables, FL, 33101-0721.
The Center for Autism Research, Department of Pediatrics, Children's Hospital of Philadelphia, University of Pennsylvania, 3535 Market Street, Suite 860, Philadelphia, PA, 19104.
Department of Psychology, Temple University, 1701 N. 13th Street, Philadelphia, PA, 19122.
Vanderbilt Kennedy Center Treatment and Research Institute for Autism Spectrum Disorders (TRIAD), Vanderbilt University, 110 Magnolia Circle, Nashville, TN, 37203.


The cost associated with incorporating standardized observational assessments and diagnostic interviews in large-scale epidemiologic studies of autism spectrum disorders (ASD) risk factors can be substantial. Streamlined approaches for confirming ASD case status would benefit these studies. We conducted a multi-site, cross-sectional criterion validity study in a convenience sample of 382 three-year olds scheduled for neurodevelopmental evaluation. ASD case classification as determined by three novel assessment instruments (the Early Video-guided Autism Screener E-VAS; the Autism Symptom Interview, ASI; the Screening Tool for Autism in Toddlers Expanded, STAT-E) each designed to be administered in less than 30 minutes by lay staff, was compared to ADOS scores and DSM-based diagnostic assessment from a qualified clinician. Sensitivity and specificity of each instrument alone and in combination were estimated. Alternative cutpoints were identified under different criteria and two-stage cross validation was used to avoid overfitting. Findings were interpreted in the context of a large, prospective pregnancy cohort study utilizing a two-stage approach to case identification. Under initial cutpoints, sensitivity ranged from 0.63 to 0.92 and specificity from 0.35 to 0.70. Cutpoints giving equal weight to sensitivity and specificity resulted in sensitivity estimates ranging from 0.45 to 0.83 and specificity ranging from 0.49 to 0.86. Several strategies were well-suited for application as a second-stage case-confirmation. These included the STAT-E alone and the parallel administration of both the E-VAS and the ASI. Use of more streamlined methods of case-confirmation in large-scale prospective cohort epidemiologic investigations of ASD risk factors appears feasible. Autism Res 2017, 10: 485-501.


ASD; autism; case-confirmation; diagnosis; epidemiology; novel assessments

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