ArticleAmerican journal on intellectual and developmental disabilities2025
Which Score for What? Operationalizing Standardized Cognitive Test Performance for the Assessment of Change.
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Article in American journal on intellectual and developmental disabilities, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.
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4citing papers in PubMed
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Who cites it
4 citing papers in PubMed.
- In-depth profile analysis and developmental trends in Wechsler performance among individuals with Williams syndrome.Child neuropsychology : a journal on normal and abnormal development in childhood and adolescence · 2026Article
- Longitudinal Characterization of Males With X-Linked Creatine Transporter Deficiency: Final Results of a Multiyear Observational Study.Pediatric neurology · 2026Observational
- Characterizing Developmental and Behavioral Profiles in Developmental Synaptopathies to Inform Clinical Trial Endpoints.American journal on intellectual and developmental disabilities · 2025Article
- A tutorial on person ability scores for the IDD clinician.International journal of developmental disabilities · 2025Article
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Authors and funding
19 authors.
Cristan FarmerCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Audrey ThurmCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Tanvi DasCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
E Martina BebinCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Jonathan A BernsteinCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Elizabeth Berry-KravisCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Joseph D BuxbaumCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Charis EngCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Thomas FrazierCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Antonio Y HardanCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Alexander KolevzonCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Darcy A KruegerCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Julian A Martinez-AgostoCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Hope NorthrupCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Craig M PowellCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Latha Valluripalli SooryaCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Joyce Y WuCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Mustafa SahinCristan Farmer, Audrey Thurm, and Tanvi Das, National Institute of Mental Health; E. Martina Bebin, University of Alabama at Birmingham; Jonathan A. Bernstein, Stanford University School of Medicine; Elizabeth Berry-Kravis, Rush University Medical Center; Joseph D. Buxbaum, Icahn School of Medicine at Mount Sinai; Charis Eng, Cleveland Clinic; Thomas Frazier, John Carroll University, SUNY Upstate Medical University; Antonio Y. Hardan, Stanford University; Alexander Kolevzon, Icahn School of Medicine at Mount Sinai; Darcy A. Krueger, Cincinnati Children's Hospital Medical Center; Julian A. Martinez-Agosto, University of California at Los Angeles; Hope Northrup, University of Texas Health Science Center at Houston, Children's Memorial Hospital; Craig M. Powell, University of Alabama at Birmingham Heersink School of Medicine and Civitan International Research Center; Latha Valluripalli Soorya, Rush University Medical Center; Joyce Y. Wu, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine; and Mustafa Sahin, Children's Hospital, Harvard Medical School.
Developmental Synaptopathies Consortium
Funding
The Impact of COVID-19 on People Living with Rare Diseases and Their FamiliesU2CTR002818 · NCATS · CINCINNATI CHILDRENS HOSP MED CTR · PI Michal Kouril · 2019 to 2026
$51.2MToward better characterization and clinical trial readiness for targeting neuropsychiatric manifestations in PTEN pathogenic variantsU54NS092090 · NINDS · BOSTON CHILDREN'S HOSPITAL · PI Darcy Krueger · 2014 to 2026
$20.1MNeurodevelopmental and Behavioral Phenotyping ZICMH002961 · NIMH · NATIONAL INSTITUTE OF MENTAL HEALTH · PI THURM, AUDREY · 2017 to 2025
$12.6MIntramural NIH HHS ZIC MH002961NCATS NIH HHS U2C TR002818NINDS NIH HHS U54 NS092090
Abstract
Developmental domains, such as cognitive, language, and motor, are key concepts of interest in longitudinal studies of intellectual and developmental disabilities (IDD). Normative scores (e.g., IQ) are often used to operationalize performance on standardized tests of these concepts, but it is the interval-distributed person-ability scores that are intended for the assessment of within-individual change. Here we illustrate the use and interpretation of several Stanford Binet, 5th Edition score types (IQ, extended IQ, Z-normalized raw score, developmental quotient, raw sum score, age equivalent, and ability score) using data from two longitudinal studies of rare genetic conditions associated with IDD. We found that, although normality assumptions were tenuous for all score types, floor effects led to model unsuitability for longitudinal analysis of most types of norm-referenced scores, and that the validity of interpretation with respect to individual change was best for ability scores.
Indexed as
Developmental DisabilitiesIntellectual DisabilityIntelligence TestsNeuropsychological TestsAdolescentAdultChildFemaleHumansIntelligenceLongitudinal StudiesMaleYoung Adultage equivalentschange sensitive scoreitem response theorylongitudinal dataperson ability scorespsychometricsrare genetic conditionsRasch analysisstandard scoresStanford-Binetsum scores
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