Evidence map›Paper›PMID 36527290›Full record

ReviewJournal of inherited metabolic disease2023

Neurocognitive and mental health impact of adrenoleukodystrophy across the lifespan: Insights for the era of newborn screening.

Elizabeth I Pierpont, Ashley R Isaia, Erin McCoy, Sarah J Brown, Ashish O Gupta, Julie B Eisengart

Open access · hybridAbstract readReview
In one paragraph

Review in Journal of inherited metabolic disease, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.

0numbers the graph read from it
0cells of the map it votes in
6citing papers in PubMed
0.7field-weighted citation impact, top 32% of its field
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

6 citing papers in PubMed, 8 citations in OpenAlex.

  1. Observational
  2. Article
  3. Article
  4. Article
  5. Article
  6. Review
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

6 authors at 1 institution in 1 country.

Elizabeth I PierpontDepartment of Pediatrics, University of Minnesota, Minneapolis, Minnesota, USA.ORCID 0000-0001-7555-8613
Ashley R IsaiaDepartment of Pediatrics, University of Minnesota, Minneapolis, Minnesota, USA.
Erin McCoyDepartment of Pediatrics, University of Minnesota, Minneapolis, Minnesota, USA.
Sarah J BrownHealth Sciences Library, University of Minnesota, Minneapolis, Minnesota, USA.
Ashish O GuptaDepartment of Pediatrics, University of Minnesota, Minneapolis, Minnesota, USA.ORCID 0000-0001-5679-5533
Julie B EisengartDepartment of Pediatrics, University of Minnesota, Minneapolis, Minnesota, USA.ORCID 0000-0001-5340-017X
University of Minnesota · US

Funding

Institutional Career Development CoreKL2TR002492 · NCATS · UNIVERSITY OF MINNESOTA · PI INGBAR, DAVID H · 2018 to 2022
$6.3M
Neurocognitive and neuroimaging markers of emerging cerebral adrenoleukodystrophyK23NS123258 · NINDS · UNIVERSITY OF MINNESOTA · PI Elizabeth Irene Pierpont · 2022 to 2026
$794k
National Institutes of Health's National Center for Advancing Translational Sciences KL2TR002492National Institutes of Health's National Institute for Neurological Disorders and Stroke K23NS123258NCATS NIH HHS KL2 TR002492NINDS NIH HHS K23 NS123258
6 · The paper itself

Abstract

X-linked adrenoleukodystrophy (ALD) is a rare inherited neurological disorder that poses considerable challenges for clinical management throughout the lifespan. Although males are generally more severely affected than females, the time course and presentation of clinical symptoms are otherwise difficult to predict. Opportunities to improve outcomes for individuals with ALD are rapidly expanding due to the introduction of newborn screening programs for this condition and an evolving treatment landscape. The aim of this comprehensive review is to synthesize current knowledge regarding the neurocognitive and mental health effects of ALD. This review provides investigators and clinicians with context to improve case conceptualization, inform prognostic counseling, and optimize neuropsychological and mental health care for patients and their families. Results highlight key predictive factors and brain-behavior relationships associated with the diverse manifestations of ALD. The review also discusses considerations for endpoints within clinical trials and identifies gaps to address in future research.

Indexed as

AdrenoleukodystrophyBrainFemaleHumansInfant, NewbornLongevityMaleMental HealthNeonatal Screeningadrenal insufficiencyadrenomyeloneuropathyassessmentbehaviorneuropsychologypsychiatric

Identifiers

PMID36527290
PMCPMC10030096
OpenAlexW4312212084

What OpenQuestion holds

Textmetadata
LicenceTDM
Read underepoch 390

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.