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

Humans Male Adrenoleukodystrophy / diagnostic imaging Diffusion Tensor Imaging Child Retrospective Studies White Matter / diagnostic imaging Corpus Callosum / diagnostic imaging Adolescent Pyramidal Tracts / diagnostic imaging Child, Preschool Hematopoietic Stem Cell Transplantation Neuropsychological Tests Cohort Studies Brain / diagnostic imaging

Journal

Neurology
ISSN: 1526-632X
Titre abrégé: Neurology
Pays: United States
ID NLM: 0401060

Informations de publication

Date de publication:
10 Sep 2024
Historique:
medline: 16 8 2024
pubmed: 16 8 2024
entrez: 16 8 2024
Statut: ppublish

Résumé

Childhood 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). In 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. Among 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; 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.

Sections du résumé

BACKGROUND AND OBJECTIVES OBJECTIVE
Childhood 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).
METHODS METHODS
In 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.
RESULTS RESULTS
Among 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 CONCLUSIONS
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.

Identifiants

DOI: 10.1212/WNL.0000000000209764 PMID: 39151102
pubmed: 39151102
doi: 10.1212/WNL.0000000000209764
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

e209764

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Auteurs

Elizabeth I Pierpont (EI)

From 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 Labounek (R)

From 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 Gupta (A)

From 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 Lund (T)

From 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 Orchard (PJ)

From 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 Dobyns (WB)

From 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 Bondy (M)

From 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 Paulson (A)

From 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 Metz (A)

From 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 Shanley (R)

From 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 Wozniak (JR)

From 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 Mueller (BA)

From 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 Loes (D)

From 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 Nascene (D)

From 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 Nestrasil (I)

From 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

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Classifications MeSH

questionsmedicales.fr › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Primates › Haplorhini › Catarrhini › Hominidae › Humains › Diffusion Tensor Imaging in Boys With...
questionsmedicales.fr › Maladies endocriniennes › Maladies des surrénales › Insuffisance surrénale › Adrénoleucodystrophie › Diffusion Tensor Imaging in Boys With...
questionsmedicales.fr › Techniques d'investigation › Neuroimagerie › Imagerie par tenseur de diffusion › Diffusion Tensor Imaging in Boys With...
questionsmedicales.fr › Personnes › Tranches d'âge › Enfant › Diffusion Tensor Imaging in Boys With...
questionsmedicales.fr › Environnement et santé publique › Santé publique › Méthodes épidémiologiques › Caractéristiques des études épidémiologiques › Études épidémiologiques › Études de cohortes › Études rétrospectives › Diffusion Tensor Imaging in Boys With...
questionsmedicales.fr › Système nerveux › Système nerveux central › Moelle spinale › Substance blanche › Diffusion Tensor Imaging in Boys With...
questionsmedicales.fr › Système nerveux › Système nerveux central › Encéphale › Prosencéphale › Télencéphale › Commissures du télencéphale › Corps calleux › Diffusion Tensor Imaging in Boys With...
questionsmedicales.fr › Personnes › Tranches d'âge › Adolescent › Diffusion Tensor Imaging in Boys With...
questionsmedicales.fr › Système nerveux › Voies nerveuses › Voies efférentes › Tractus pyramidaux › Diffusion Tensor Imaging in Boys With...
questionsmedicales.fr › Personnes › Tranches d'âge › Enfant › Enfant d'âge préscolaire › Diffusion Tensor Imaging in Boys With...
questionsmedicales.fr › Procédures de chirurgie opératoire › Transplantation › Transplantation cellulaire › Transplantation de cellules souches › Transplantation de cellules souches hématopoïétiques › Diffusion Tensor Imaging in Boys With...
questionsmedicales.fr › Disciplines et activités comportementales › Tests psychologiques › Tests neuropsychologiques › Diffusion Tensor Imaging in Boys With...
questionsmedicales.fr › Environnement et santé publique › Santé publique › Méthodes épidémiologiques › Caractéristiques des études épidémiologiques › Études épidémiologiques › Études de cohortes › Diffusion Tensor Imaging in Boys With...
questionsmedicales.fr › Système nerveux › Système nerveux central › Encéphale › Diffusion Tensor Imaging in Boys With...