Evidence map›Paper›PMID 42040243›Full record

ReviewNeurology. Genetics2026

Expert Recommendations for Rapid Response to Positive Newborn Screen for Infantile Krabbe Disease.

Robert Thompson Stone, Amy L White, Jennifer P Rubin, Deepa Soundara Rajan, Dietrich Matern, Joanne Kurtzberg, Joseph J Orsini, Shalini Shenoy, Eric J Mallack, Margie A Ream

Abstract readReview
In one paragraph

Review in Neurology. Genetics, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing 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

0 citing papers in PubMed.

No citing paper in PubMed yet.

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.

Robert Thompson StoneGolisano Children's Hospital, University of Rochester, NY.ORCID https://orcid.org/0000-0003-1062-854X
Amy L WhiteMayo Clinic, Rochester, MN.ORCID https://orcid.org/0000-0001-8226-615X
Jennifer P RubinAnn & Robert H. Lurie Children's Hospital of Chicago, Northwestern University, IL.ORCID https://orcid.org/0000-0001-5594-3389
Deepa Soundara RajanUPMC Children's Hospital of Pittsburgh, University of Pittsburgh, PA.ORCID https://orcid.org/0000-0001-5699-8541
Dietrich MaternMayo Clinic, Rochester, MN.ORCID https://orcid.org/0000-0001-6298-4138
Joanne KurtzbergDuke Children's, Duke University, Durham, NC.ORCID https://orcid.org/0000-0002-3370-0703
Joseph J OrsiniWadsworth Center, New York State Department of Health, Albany.ORCID https://orcid.org/0000-0002-7081-6027
Shalini ShenoySt. Louis Children's Hospital, Washington University, MO.ORCID https://orcid.org/0000-0002-9283-607X
Eric J MallackKennedy Krieger Institute, Johns Hopkins University, Baltimore, MD; and.ORCID https://orcid.org/0000-0002-2678-8807
Margie A ReamNationwide Children's Hospital, The Ohio State University, Columbus.ORCID https://orcid.org/0000-0001-5749-3297

Funding

Sleep and Circadian Dysfunction, Brain and Neurobehavioral Development in AutismP50HD103538 · NICHD · HUGO W. MOSER RES INST KENNEDY KRIEGER · PI S. Ali Fatemi, Bradley L Schlaggar · 2020 to 2026
$9.9M
NICHD NIH HHS P50 HD103538
6 · The paper itself

Abstract

Infantile Krabbe disease (IKD) is a rapidly progressive leukodystrophy for which hematopoietic stem cell transplantation (HCT) offers the only disease-modifying treatment. HCT must occur presymptomatically, ideally by 30 days of life, to optimize survival and neurodevelopmental outcomes. With the expansion of newborn screening for Krabbe disease (KD), pediatric neurologists and geneticists will be responsible for obtaining urgent diagnostic testing and connecting families to experts for information and treatment. Given the variability in state-specific screening protocols, clinicians must be familiar with their local algorithms to act quickly on receiving abnormal results and refer to nearby, specialty treatment centers. Informed consent discussions must clearly outline the natural history of untreated IKD, the requirement for rapid, presymptomatic treatment, and provide a realistic view of the benefits and limitations of HCT. Supportive and palliative care should be prioritized regardless of treatment choice. This publication provides expert guidance on interpreting newborn screening results, conducting rapid confirmatory testing, and referring to and/or establishing treatment centers ready to care for IKD. With anticipatory planning and interdisciplinary collaboration, pediatric specialists can provide accurate diagnosis, compassionate counseling, and expedited evaluation to meet the narrow window for effective HCT.

Identifiers

PMID42040243
PMCPMC13105195

What OpenQuestion holds

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LicenceCC BY-NC-ND
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Registered trials

None linked

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.