Evidence map›Paper›PMID 39438712›Full record

ReviewPediatric research2025

Early Intervention services in the era of genomic medicine: setting a research agenda.

Katherine E MacDuffie, Betty Cohn, Paul Appelbaum, Kyle B Brothers, Dan Doherty, Aaron J Goldenberg, Elizabeth Reynolds, Hadley Stevens Smith, Anne Wheeler, Joon-Ho Yu

Abstract readReview
In one paragraph

Review in Pediatric research, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

4 citing papers in PubMed.

  1. Article
  2. Article
  3. Article
  4. Parent Reports of Developmental Service Utilization After Newborn Screening.International journal of neonatal screening · 2024
    Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

10 authors.

Katherine E MacDuffieTreuman Katz Center for Pediatric Bioethics and Palliative Care, Seattle Children's Research Institute, Seattle, WA, USA. kate.macduffie@seattlechildrens.org.
Betty CohnInstitute for Public Health Genetics, University of Washington, Seattle, WA, USA.
Paul AppelbaumDepartment of Psychiatry, Columbia University Irving Medical Center and NY State Psychiatric Institute, New York, NY, USA.
Kyle B BrothersDepartment of Pediatrics, University of Louisville, Louisville, KY, USA.
Dan DohertyDepartment of Pediatrics, University of Washington School of Medicine, Seattle, WA, USA.
Aaron J GoldenbergDepartment of Bioethics, Case Western Reserve University School of Medicine, Cleveland, OH, USA.
Elizabeth ReynoldsRTI International, Research Triangle Park, NC, USA.
Hadley Stevens SmithPrecision Medicine Translational Research (PROMoTeR) Center, Department of Population Medicine, Harvard Medical School and Harvard Pilgrim Health Care Institute, Boston, MA, USA.
Anne WheelerRTI International, Research Triangle Park, NC, USA.
Joon-Ho YuTreuman Katz Center for Pediatric Bioethics and Palliative Care, Seattle Children's Research Institute, Seattle, WA, USA.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Newborn genomic sequencing (NBSeq) has the potential to substantially improve early detection of rare genetic conditions, allowing for pre-symptomatic treatment to optimize outcomes. Expanding conceptions of the clinical utility of NBSeq include earlier access to behavioral early intervention to support the acquisition of core motor, cognitive, communication, and adaptive skills during critical windows in early development. However, important questions remain about equitable access to early intervention programs for the growing number of infants identified with a genetic condition via NBSeq. We review the current NBSeq public health, clinical, and research landscape, and highlight ongoing international research efforts to collect population-level data on the utility of NBSeq for healthy newborns. We then explore the challenges facing a specific Early Intervention (EI) system-the US federally supported "Part C" system-for meeting the developmental needs of young children with genetic diagnoses, including structural limitations related to funding, variable eligibility criteria, and lack of collaboration with newborn screening programs. We conclude with a set of questions to guide future research at the intersection of NBSeq, newborn screening, and EI, which once answered, can steer future policy to ensure that EI service systems can optimally support the developmental needs of infants impacted by broader implementation of NBSeq. IMPACT: Existing literature on the clinical benefits of genome sequencing in newborns tends to focus on earlier provision of medical interventions, with less attention to the ongoing developmental needs of very young children with genetic conditions. This review outlines the developmental needs of a growing number of children diagnosed with genetic conditions in infancy and describes the strengths and limitations of the United States Early Intervention system (IDEA Part C) for meeting those needs.

Indexed as

Early Intervention, EducationalEarly Medical InterventionGenetic Diseases, InbornGenomicsNeonatal ScreeningGenetic TestingHumansInfantInfant, NewbornUnited States

Identifiers

PMID39438712
PMCPMC12106093

What OpenQuestion holds

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.