Risk factor analysis of cerebral white matter hyperintensities in children with sickle cell disease
Open accessAbstract
Summary: Sickle cell disease (SCD) is complicated by silent cerebral infarcts, visible as white matter hyperintensities (WMH s) on magnetic resonance imaging (MRI). Both local vaso‐occlusion, elicited by endothelial dysfunction, and insufficiency of cerebral blood flow (CBF) have been proposed to be involved in the aetiology. We performed an explorative study to investigate the associations between WMH s and markers of endothelial dysfunction and CBF by quantifying WMH volume on 3·0 Tesla MRI. We included 40 children with Hb SS or HbSβ 0 thalassaemia, with a mean age of 12·1 ± 2·6 years. Boys demonstrated an increased risk for WMH s (odds ratio 4·5, 95% confidence interval 1·2–17·4), unrelated to glucose‐6‐phosphate dehydrogenase deficiency. In patients with WMH s, lower fetal haemoglobin (HbF) was associated with a larger WMH volume (regression coefficient = −0·62, R2 = 0·25, P = 0·04). Lower ADAMTS 13 (a disintegrin and metalloproteinase with a thrombospondin type 1 motif, member 13) levels were associated with lower CBF in the white matter (regression coefficient = 0·07, R2 = 0·15, P = 0·03), suggesting that endothelial dysfunction could potentially hamper CBF. The findings of our explorative study suggest that a high level of HbF may be protective for WMH s and that endothelial dysfunction may contribute to the development of WMH s by reducing CBF.
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Children aged 12.1±2.6 years with the highest HbF were protected from silent infarctions.
“Children aged 12.1 6 2.6 years with the highest HbF were protected from silent infarctions.68”
From Fetal hemoglobin in sickle cell anemia (2020) · cites this paper as [68]
The Abstract reports a mean age of 12.1 ± 2.6 years and suggests that high HbF may be protective for white matter hyperintensities. The Results specify a narrower finding: among children who already had these lesions, lower HbF was associated with greater lesion volume. The paper does not establish that children with the highest HbF were protected from developing silent infarctions. (LLM, conf=0.89)
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Quantitative susceptibility mapping (QSM) and R2* of silent cerebral infarcts in sickle cell anemia
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Self-citation
- Authors
- van der Land, V., Mutsaerts, H. J. M. M., Engelen, M., Heijboer, H., Roest, M., Hollestelle, M. J., Kuijpers, T. W., Nederkoorn, P. J., Cnossen, M. H., Majoie, C. B. L. M., Nederveen, A. J., & Fijnvandraat, K.
- Affiliations
- Department of Paediatric Haematology, Immunology and Infectious Diseases Emma Children's Hospital Academic Medical Centre Amsterdam The Netherlands
- Department of Radiology Academic Medical Centre Amsterdam The Netherlands
- Amsterdam UMC Location University of Amsterdam (NL)
- University Medical Center Utrecht (NL)
- Sanquin (NL)
- Erasmus MC (NL)
- Corresponding author
- Fijnvandraat, Karin
- ORCID
- Venue
- British Journal of Haematology
- Publisher
- Wiley
- Published
- 2015-10-22
- Volume
- 172
- Issue
- 2
- Pages
- 274-284
- ISSN
- 0007-1048 (ISSN-L)
- 1365-2141
- OpenAlex
- W2194025732
- Semantic Scholar
- 58bdf98376d66f63836655dc8f22cbf7ddfb35db
- Cited by
- 30
- References
- 60
- Citations by year
- 2025: 2 · 2024: 1 · 2022: 4 · 2021: 4 · 2020: 8 · 2019: 1 · 2018: 1 · 2017: 5 · 2016: 4
- FWCI
- 1.87
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Locations
- British Journal of Haematology (publishedVersion)
- PubMed (publishedVersion)
- Pure Amsterdam UMC (submittedVersion, other-oa)
- Data Archiving and Networked Services (DANS) (submittedVersion)
- EUR Research Repository (Erasmus University Rotterdam) (submittedVersion)
- Amsterdam UMC (VU Amsterdam) - Institutional Repository (submittedVersion)
- Funders
- Fonds Nuts Ohra, the Netherlands
- Topics
- Health Sciences › Medicine › Genetics › Hemoglobinopathies and Related Disorders
- Health Sciences › Medicine › Radiology, Nuclear Medicine and Imaging › Advanced MRI Techniques and Applications
- Health Sciences › Medicine › Epidemiology › Acute Ischemic Stroke Management
- Keywords
- sickle cell disease, white matter hyperintensities, silent cerebral infarction, endothelial dysfunction, cerebral blood flow, ADAMTS13, fetal hemoglobin, children, risk factors, vascular occlusion
- MeSH terms
- Adolescent, Anemia, Sickle Cell, Blood Coagulation, Cerebral Infarction, Cerebrovascular Circulation, Child, Endothelium, Vascular, Female, Fetal Hemoglobin, Humans, Magnetic Resonance Imaging, Male, Prospective Studies, Risk Factors, Sex Factors, White Matter
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