Evidence map›Paper›PMID 39151102›Full record

ArticleNeurology2024

Diffusion Tensor Imaging in Boys With Adrenoleukodystrophy: Identification of Cerebral Disease and Association With Neurocognitive Outcomes.

Elizabeth I Pierpont, René Labounek, Ashish Gupta, Troy Lund, Paul J Orchard, William B Dobyns, Monica Bondy, Amy Paulson, Andrew Metz, Ryan Shanley and 5 more

Abstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Neurology, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 5 papers.

0numbers the graph read from it
0cells of the map it votes in
5citing 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

5 citing papers in PubMed.

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

15 authors.

Elizabeth I PierpontFrom the Departments of Pediatrics (E.I.P., R.L., A.G., T.L., P.J.O., W.B.D., M.B., A.P., I.N.), Neurology (A.M.), Psychiatry & Behavioral Sciences (J.R.W., B.A.M.), and Radiology (D.N.), University of Minnesota Medical School, Minneapolis; Biostatistical Design and Analysis Center (R.S.), Clinical and Translational Science Institute, University of Minnesota, Minneapolis; and Independent Neuroradiologist-Consultant (D.L.), Minneapolis, MN.ORCID 0000-0001-7555-8613
René LabounekFrom the Departments of Pediatrics (E.I.P., R.L., A.G., T.L., P.J.O., W.B.D., M.B., A.P., I.N.), Neurology (A.M.), Psychiatry & Behavioral Sciences (J.R.W., B.A.M.), and Radiology (D.N.), University of Minnesota Medical School, Minneapolis; Biostatistical Design and Analysis Center (R.S.), Clinical and Translational Science Institute, University of Minnesota, Minneapolis; and Independent Neuroradiologist-Consultant (D.L.), Minneapolis, MN.
Ashish GuptaFrom the Departments of Pediatrics (E.I.P., R.L., A.G., T.L., P.J.O., W.B.D., M.B., A.P., I.N.), Neurology (A.M.), Psychiatry & Behavioral Sciences (J.R.W., B.A.M.), and Radiology (D.N.), University of Minnesota Medical School, Minneapolis; Biostatistical Design and Analysis Center (R.S.), Clinical and Translational Science Institute, University of Minnesota, Minneapolis; and Independent Neuroradiologist-Consultant (D.L.), Minneapolis, MN.ORCID 0000-0001-5679-5533
Troy LundFrom the Departments of Pediatrics (E.I.P., R.L., A.G., T.L., P.J.O., W.B.D., M.B., A.P., I.N.), Neurology (A.M.), Psychiatry & Behavioral Sciences (J.R.W., B.A.M.), and Radiology (D.N.), University of Minnesota Medical School, Minneapolis; Biostatistical Design and Analysis Center (R.S.), Clinical and Translational Science Institute, University of Minnesota, Minneapolis; and Independent Neuroradiologist-Consultant (D.L.), Minneapolis, MN.ORCID 0000-0001-9174-8919
Paul J OrchardFrom the Departments of Pediatrics (E.I.P., R.L., A.G., T.L., P.J.O., W.B.D., M.B., A.P., I.N.), Neurology (A.M.), Psychiatry & Behavioral Sciences (J.R.W., B.A.M.), and Radiology (D.N.), University of Minnesota Medical School, Minneapolis; Biostatistical Design and Analysis Center (R.S.), Clinical and Translational Science Institute, University of Minnesota, Minneapolis; and Independent Neuroradiologist-Consultant (D.L.), Minneapolis, MN.
William B DobynsFrom the Departments of Pediatrics (E.I.P., R.L., A.G., T.L., P.J.O., W.B.D., M.B., A.P., I.N.), Neurology (A.M.), Psychiatry & Behavioral Sciences (J.R.W., B.A.M.), and Radiology (D.N.), University of Minnesota Medical School, Minneapolis; Biostatistical Design and Analysis Center (R.S.), Clinical and Translational Science Institute, University of Minnesota, Minneapolis; and Independent Neuroradiologist-Consultant (D.L.), Minneapolis, MN.
Monica BondyFrom the Departments of Pediatrics (E.I.P., R.L., A.G., T.L., P.J.O., W.B.D., M.B., A.P., I.N.), Neurology (A.M.), Psychiatry & Behavioral Sciences (J.R.W., B.A.M.), and Radiology (D.N.), University of Minnesota Medical School, Minneapolis; Biostatistical Design and Analysis Center (R.S.), Clinical and Translational Science Institute, University of Minnesota, Minneapolis; and Independent Neuroradiologist-Consultant (D.L.), Minneapolis, MN.
Amy PaulsonFrom the Departments of Pediatrics (E.I.P., R.L., A.G., T.L., P.J.O., W.B.D., M.B., A.P., I.N.), Neurology (A.M.), Psychiatry & Behavioral Sciences (J.R.W., B.A.M.), and Radiology (D.N.), University of Minnesota Medical School, Minneapolis; Biostatistical Design and Analysis Center (R.S.), Clinical and Translational Science Institute, University of Minnesota, Minneapolis; and Independent Neuroradiologist-Consultant (D.L.), Minneapolis, MN.
Andrew MetzFrom the Departments of Pediatrics (E.I.P., R.L., A.G., T.L., P.J.O., W.B.D., M.B., A.P., I.N.), Neurology (A.M.), Psychiatry & Behavioral Sciences (J.R.W., B.A.M.), and Radiology (D.N.), University of Minnesota Medical School, Minneapolis; Biostatistical Design and Analysis Center (R.S.), Clinical and Translational Science Institute, University of Minnesota, Minneapolis; and Independent Neuroradiologist-Consultant (D.L.), Minneapolis, MN.
Ryan ShanleyFrom the Departments of Pediatrics (E.I.P., R.L., A.G., T.L., P.J.O., W.B.D., M.B., A.P., I.N.), Neurology (A.M.), Psychiatry & Behavioral Sciences (J.R.W., B.A.M.), and Radiology (D.N.), University of Minnesota Medical School, Minneapolis; Biostatistical Design and Analysis Center (R.S.), Clinical and Translational Science Institute, University of Minnesota, Minneapolis; and Independent Neuroradiologist-Consultant (D.L.), Minneapolis, MN.ORCID 0000-0003-2398-5466
Jeffrey R WozniakFrom the Departments of Pediatrics (E.I.P., R.L., A.G., T.L., P.J.O., W.B.D., M.B., A.P., I.N.), Neurology (A.M.), Psychiatry & Behavioral Sciences (J.R.W., B.A.M.), and Radiology (D.N.), University of Minnesota Medical School, Minneapolis; Biostatistical Design and Analysis Center (R.S.), Clinical and Translational Science Institute, University of Minnesota, Minneapolis; and Independent Neuroradiologist-Consultant (D.L.), Minneapolis, MN.
Bryon A MuellerFrom the Departments of Pediatrics (E.I.P., R.L., A.G., T.L., P.J.O., W.B.D., M.B., A.P., I.N.), Neurology (A.M.), Psychiatry & Behavioral Sciences (J.R.W., B.A.M.), and Radiology (D.N.), University of Minnesota Medical School, Minneapolis; Biostatistical Design and Analysis Center (R.S.), Clinical and Translational Science Institute, University of Minnesota, Minneapolis; and Independent Neuroradiologist-Consultant (D.L.), Minneapolis, MN.
Daniel LoesFrom the Departments of Pediatrics (E.I.P., R.L., A.G., T.L., P.J.O., W.B.D., M.B., A.P., I.N.), Neurology (A.M.), Psychiatry & Behavioral Sciences (J.R.W., B.A.M.), and Radiology (D.N.), University of Minnesota Medical School, Minneapolis; Biostatistical Design and Analysis Center (R.S.), Clinical and Translational Science Institute, University of Minnesota, Minneapolis; and Independent Neuroradiologist-Consultant (D.L.), Minneapolis, MN.
David NasceneFrom the Departments of Pediatrics (E.I.P., R.L., A.G., T.L., P.J.O., W.B.D., M.B., A.P., I.N.), Neurology (A.M.), Psychiatry & Behavioral Sciences (J.R.W., B.A.M.), and Radiology (D.N.), University of Minnesota Medical School, Minneapolis; Biostatistical Design and Analysis Center (R.S.), Clinical and Translational Science Institute, University of Minnesota, Minneapolis; and Independent Neuroradiologist-Consultant (D.L.), Minneapolis, MN.
Igor NestrasilFrom the Departments of Pediatrics (E.I.P., R.L., A.G., T.L., P.J.O., W.B.D., M.B., A.P., I.N.), Neurology (A.M.), Psychiatry & Behavioral Sciences (J.R.W., B.A.M.), and Radiology (D.N.), University of Minnesota Medical School, Minneapolis; Biostatistical Design and Analysis Center (R.S.), Clinical and Translational Science Institute, University of Minnesota, Minneapolis; and Independent Neuroradiologist-Consultant (D.L.), Minneapolis, MN.ORCID 0000-0002-3833-1484

Funding

Neurocognitive and neuroimaging markers of emerging cerebral adrenoleukodystrophyK23NS123258 · NINDS · UNIVERSITY OF MINNESOTA · PI Elizabeth Irene Pierpont · 2022 to 2026
$794k
NINDS NIH HHS K23 NS123258
6 · The paper itself

Abstract

BACKGROUND AND

objectivesChildhood cerebral adrenoleukodystrophy (C-ALD) is a severe inflammatory demyelinating disease that must be treated at an early stage to prevent permanent brain injury and neurocognitive decline. In standard clinical practice, C-ALD lesions are detected and characterized by a neuroradiologist reviewing anatomical MRI scans. We aimed to assess whether diffusion tensor imaging (DTI) is sensitive to the presence and severity of C-ALD lesions and to investigate associations with neurocognitive outcomes after hematopoietic cell therapy (HCT).

methodsIn this retrospective cohort study, we analyzed high-resolution anatomical MRI, DTI, and neurocognitive assessments from boys with C-ALD undergoing HCT at the University of Minnesota between 2011 and 2021. Longitudinal DTI data were compared with an age-matched group of boys with ALD and no lesion (NL-ALD). DTI metrics were obtained for atlas-based regions of interest (ROIs) within 3 subdivisions of the corpus callosum (CC), corticospinal tract (CST), and total white matter (WM). Between-group baseline and slope differences in fractional anisotropy (FA) and axial (AD), radial (RD), and mean (MD) diffusivities were compared using analysis of covariance accounting for age, MRI severity (Loes score), and lesion location.

resultsAmong patients with NL-ALD (n = 14), stable or increasing FA, stable AD, and stable or decreasing RD and MD were generally observed during the 1-year study period across all ROIs. In comparison, patients with mild posterior lesions (Loes 1-2; n = 13) demonstrated lower baseline FA in the CC splenium (C-ALD 0.50 ± 0.08 vs NL-ALD 0.58 ± 0.04; DISCUSSION: DTI was sensitive to lesion presence and severity as well as clinical neurocognitive effects of C-ALD. DTI metrics quantify C-ALD even at an early stage.

Indexed as

AdrenoleukodystrophyCorpus CallosumDiffusion Tensor ImagingWhite MatterAdolescentBrainChildChild, PreschoolCohort StudiesHematopoietic Stem Cell TransplantationHumansMaleNeuropsychological TestsPyramidal TractsRetrospective Studies

Identifiers

PMID39151102
PMCPMC11329293

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
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.