Evidence map›Paper›PMID 39546738›Full record

ArticleNeurology2024

Cerebral Oxygen Metabolic Stress in Children and Adults With Large Vessel Vasculopathy Due to Sickle Cell Disease.

Yan Wang, Slim Fellah, Martin Reis, Kristin P Guilliams, Melanie E Fields, Karen Steger-May, Amy E Mirro, Josiah B Lewis, Chunwei Ying, Rachel A Cohen and 6 more

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Article in Neurology, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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5 · Who and what money

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16 authors.

Yan WangFrom the Department of Neurology (Y.W., S.F., K.G., M.E.F., J.B.L., Y.C., J.-M.L.), Mallinckrodt Institute of Radiology (M.R., K.G., M.E.F., C.Y., J.-M.L., H.A.), and Division of Pediatrics (K.G., A.E.M., M.L.H.), Center for Biostatistics and Data Science (K.S.-M.), Washington University School of Medicine; Washington University in St. Louis (R.A.C.); and Division of Hematology/Oncology (A.A.K., A.L.F.), Department of Medicine, Washington University School of Medicine, St. Louis, MO.ORCID 0000-0002-3750-0311
Slim FellahFrom the Department of Neurology (Y.W., S.F., K.G., M.E.F., J.B.L., Y.C., J.-M.L.), Mallinckrodt Institute of Radiology (M.R., K.G., M.E.F., C.Y., J.-M.L., H.A.), and Division of Pediatrics (K.G., A.E.M., M.L.H.), Center for Biostatistics and Data Science (K.S.-M.), Washington University School of Medicine; Washington University in St. Louis (R.A.C.); and Division of Hematology/Oncology (A.A.K., A.L.F.), Department of Medicine, Washington University School of Medicine, St. Louis, MO.ORCID 0000-0001-6179-6389
Martin ReisFrom the Department of Neurology (Y.W., S.F., K.G., M.E.F., J.B.L., Y.C., J.-M.L.), Mallinckrodt Institute of Radiology (M.R., K.G., M.E.F., C.Y., J.-M.L., H.A.), and Division of Pediatrics (K.G., A.E.M., M.L.H.), Center for Biostatistics and Data Science (K.S.-M.), Washington University School of Medicine; Washington University in St. Louis (R.A.C.); and Division of Hematology/Oncology (A.A.K., A.L.F.), Department of Medicine, Washington University School of Medicine, St. Louis, MO.
Kristin P GuilliamsFrom the Department of Neurology (Y.W., S.F., K.G., M.E.F., J.B.L., Y.C., J.-M.L.), Mallinckrodt Institute of Radiology (M.R., K.G., M.E.F., C.Y., J.-M.L., H.A.), and Division of Pediatrics (K.G., A.E.M., M.L.H.), Center for Biostatistics and Data Science (K.S.-M.), Washington University School of Medicine; Washington University in St. Louis (R.A.C.); and Division of Hematology/Oncology (A.A.K., A.L.F.), Department of Medicine, Washington University School of Medicine, St. Louis, MO.ORCID 0000-0002-5518-3778
Melanie E FieldsFrom the Department of Neurology (Y.W., S.F., K.G., M.E.F., J.B.L., Y.C., J.-M.L.), Mallinckrodt Institute of Radiology (M.R., K.G., M.E.F., C.Y., J.-M.L., H.A.), and Division of Pediatrics (K.G., A.E.M., M.L.H.), Center for Biostatistics and Data Science (K.S.-M.), Washington University School of Medicine; Washington University in St. Louis (R.A.C.); and Division of Hematology/Oncology (A.A.K., A.L.F.), Department of Medicine, Washington University School of Medicine, St. Louis, MO.ORCID 0000-0001-6223-4468
Karen Steger-MayFrom the Department of Neurology (Y.W., S.F., K.G., M.E.F., J.B.L., Y.C., J.-M.L.), Mallinckrodt Institute of Radiology (M.R., K.G., M.E.F., C.Y., J.-M.L., H.A.), and Division of Pediatrics (K.G., A.E.M., M.L.H.), Center for Biostatistics and Data Science (K.S.-M.), Washington University School of Medicine; Washington University in St. Louis (R.A.C.); and Division of Hematology/Oncology (A.A.K., A.L.F.), Department of Medicine, Washington University School of Medicine, St. Louis, MO.
Amy E MirroFrom the Department of Neurology (Y.W., S.F., K.G., M.E.F., J.B.L., Y.C., J.-M.L.), Mallinckrodt Institute of Radiology (M.R., K.G., M.E.F., C.Y., J.-M.L., H.A.), and Division of Pediatrics (K.G., A.E.M., M.L.H.), Center for Biostatistics and Data Science (K.S.-M.), Washington University School of Medicine; Washington University in St. Louis (R.A.C.); and Division of Hematology/Oncology (A.A.K., A.L.F.), Department of Medicine, Washington University School of Medicine, St. Louis, MO.ORCID 0000-0001-5501-7342
Josiah B LewisFrom the Department of Neurology (Y.W., S.F., K.G., M.E.F., J.B.L., Y.C., J.-M.L.), Mallinckrodt Institute of Radiology (M.R., K.G., M.E.F., C.Y., J.-M.L., H.A.), and Division of Pediatrics (K.G., A.E.M., M.L.H.), Center for Biostatistics and Data Science (K.S.-M.), Washington University School of Medicine; Washington University in St. Louis (R.A.C.); and Division of Hematology/Oncology (A.A.K., A.L.F.), Department of Medicine, Washington University School of Medicine, St. Louis, MO.ORCID 0000-0001-5758-245X
Chunwei YingFrom the Department of Neurology (Y.W., S.F., K.G., M.E.F., J.B.L., Y.C., J.-M.L.), Mallinckrodt Institute of Radiology (M.R., K.G., M.E.F., C.Y., J.-M.L., H.A.), and Division of Pediatrics (K.G., A.E.M., M.L.H.), Center for Biostatistics and Data Science (K.S.-M.), Washington University School of Medicine; Washington University in St. Louis (R.A.C.); and Division of Hematology/Oncology (A.A.K., A.L.F.), Department of Medicine, Washington University School of Medicine, St. Louis, MO.ORCID 0000-0003-1060-3289
Rachel A CohenFrom the Department of Neurology (Y.W., S.F., K.G., M.E.F., J.B.L., Y.C., J.-M.L.), Mallinckrodt Institute of Radiology (M.R., K.G., M.E.F., C.Y., J.-M.L., H.A.), and Division of Pediatrics (K.G., A.E.M., M.L.H.), Center for Biostatistics and Data Science (K.S.-M.), Washington University School of Medicine; Washington University in St. Louis (R.A.C.); and Division of Hematology/Oncology (A.A.K., A.L.F.), Department of Medicine, Washington University School of Medicine, St. Louis, MO.ORCID 0009-0009-3722-1468
Monica L HulbertFrom the Department of Neurology (Y.W., S.F., K.G., M.E.F., J.B.L., Y.C., J.-M.L.), Mallinckrodt Institute of Radiology (M.R., K.G., M.E.F., C.Y., J.-M.L., H.A.), and Division of Pediatrics (K.G., A.E.M., M.L.H.), Center for Biostatistics and Data Science (K.S.-M.), Washington University School of Medicine; Washington University in St. Louis (R.A.C.); and Division of Hematology/Oncology (A.A.K., A.L.F.), Department of Medicine, Washington University School of Medicine, St. Louis, MO.
Allison A KingFrom the Department of Neurology (Y.W., S.F., K.G., M.E.F., J.B.L., Y.C., J.-M.L.), Mallinckrodt Institute of Radiology (M.R., K.G., M.E.F., C.Y., J.-M.L., H.A.), and Division of Pediatrics (K.G., A.E.M., M.L.H.), Center for Biostatistics and Data Science (K.S.-M.), Washington University School of Medicine; Washington University in St. Louis (R.A.C.); and Division of Hematology/Oncology (A.A.K., A.L.F.), Department of Medicine, Washington University School of Medicine, St. Louis, MO.ORCID 0000-0002-1951-6176
Yasheng ChenFrom the Department of Neurology (Y.W., S.F., K.G., M.E.F., J.B.L., Y.C., J.-M.L.), Mallinckrodt Institute of Radiology (M.R., K.G., M.E.F., C.Y., J.-M.L., H.A.), and Division of Pediatrics (K.G., A.E.M., M.L.H.), Center for Biostatistics and Data Science (K.S.-M.), Washington University School of Medicine; Washington University in St. Louis (R.A.C.); and Division of Hematology/Oncology (A.A.K., A.L.F.), Department of Medicine, Washington University School of Medicine, St. Louis, MO.
Jin-Moo LeeFrom the Department of Neurology (Y.W., S.F., K.G., M.E.F., J.B.L., Y.C., J.-M.L.), Mallinckrodt Institute of Radiology (M.R., K.G., M.E.F., C.Y., J.-M.L., H.A.), and Division of Pediatrics (K.G., A.E.M., M.L.H.), Center for Biostatistics and Data Science (K.S.-M.), Washington University School of Medicine; Washington University in St. Louis (R.A.C.); and Division of Hematology/Oncology (A.A.K., A.L.F.), Department of Medicine, Washington University School of Medicine, St. Louis, MO.ORCID 0000-0002-3979-0906
Hongyu AnFrom the Department of Neurology (Y.W., S.F., K.G., M.E.F., J.B.L., Y.C., J.-M.L.), Mallinckrodt Institute of Radiology (M.R., K.G., M.E.F., C.Y., J.-M.L., H.A.), and Division of Pediatrics (K.G., A.E.M., M.L.H.), Center for Biostatistics and Data Science (K.S.-M.), Washington University School of Medicine; Washington University in St. Louis (R.A.C.); and Division of Hematology/Oncology (A.A.K., A.L.F.), Department of Medicine, Washington University School of Medicine, St. Louis, MO.ORCID 0000-0001-6459-2269
Andria L FordFrom the Department of Neurology (Y.W., S.F., K.G., M.E.F., J.B.L., Y.C., J.-M.L.), Mallinckrodt Institute of Radiology (M.R., K.G., M.E.F., C.Y., J.-M.L., H.A.), and Division of Pediatrics (K.G., A.E.M., M.L.H.), Center for Biostatistics and Data Science (K.S.-M.), Washington University School of Medicine; Washington University in St. Louis (R.A.C.); and Division of Hematology/Oncology (A.A.K., A.L.F.), Department of Medicine, Washington University School of Medicine, St. Louis, MO.ORCID 0000-0003-0605-8943

Funding

The Impact of Cerebral Metabolic Stress on the Development of the Structural and Functional Connectome in Pediatric Sickle Cell AnemiaR01HL157188 · NHLBI · WASHINGTON UNIVERSITY · PI FIELDS, MELANIE ERIN · 2021 to 2025
$3.1M
NHLBI NIH HHS R01 HL157188
6 · The paper itself

Abstract

BACKGROUND AND

objectivesLarge vessel vasculopathy (LVV), or moyamoya syndrome, increases the risk of stroke in patients with sickle cell disease (SCD), yet effective treatments are lacking. In atherosclerotic carotid disease, previous studies demonstrated elevated oxygen extraction fraction (OEF) as a predictor of ipsilateral stroke. In a SCD cohort, we examined hemispheric hemodynamic and oxygen metabolic dysfunction as tissue-based biomarkers of cerebral ischemic risk in patients with LVV.

methodsChildren and adults with SCD were recruited from a SCD clinic associated with a tertiary medical center and underwent prospective brain MRI and MR angiography. LVV was defined as ≥75% stenosis in a major anterior circulation artery, excluding occlusion or previous revascularization surgery. Baseline characteristics, cerebral blood flow (CBF), normalized OEF (nOEF), infarct volume, white matter microstructure, and brain volume were compared in hemispheres with vs without LVV. In a cross-sectional analysis, mixed-effects linear multivariable models examined the effect of LVV on: (1) CBF and nOEF, as tissue markers of hemodynamic and oxygen metabolic stress, respectively, and (2) endpoints of cerebral ischemic injury including infarct volume, white matter microstructure, and brain volume.

resultsOf 155 patients (22 [12-31] years, 57% female), 33 (21%) had ≥25% stenosis, 22 (14%) had ≥50% stenosis, 14 (9%) had 75%-99% stenosis, and 5 (3%) had 100% occlusion. After excluding hemispheres with previous revascularization surgery, LVV was present in 16 hemispheres from 11 patients. Hemispheres with (N = 16) vs without (N = 283) LVV had lower CBF (25.2 vs 32.1 mL/100 g/min, DISCUSSION: Beyond greater infarct burden, LVV was associated with hemispheric atrophy and white matter microstructural injury. As an indicator of active hypoxia, elevated nOEF likely represents a compensatory response to flow-limiting stenosis in hemispheres with LVV. The study is limited by a small number of patients with severe stenosis. Future studies are needed to evaluate the potential of tissue-based CBF and nOEF in assessing stroke risk and guide timely treatment of vasculopathy in SCD.

Indexed as

Anemia, Sickle CellCerebrovascular CirculationMoyamoya DiseaseAdolescentAdultBrainChildCross-Sectional StudiesFemaleHumansMagnetic Resonance AngiographyMagnetic Resonance ImagingMaleOxygenProspective StudiesYoung AdultOxygen

Identifiers

PMID39546738
PMCPMC11573263

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