Intracranial atherosclerosis in pseudoxanthoma elasticum : A case-control study
Copyright © 2022 The Authors. Published by Elsevier B.V. All rights reserved..
BACKGROUND AND AIMS: Pseudoxanthoma elasticum (PXE) is a genetic disorder characterized by systemic calcification of elastin fibers. Additionally, PXE is associated with an increased risk of stroke. It has been hypothesized that this may be caused by accelerated (intracranial) atherogenesis, as a consequence of specific genetic mutations underlying PXE. Hence, we compared the distribution and burden of intracranial atherosclerosis between PXE patients and healthy controls.
METHODS: Fifty PXE patients and 40 age-and-sex-matched healthy controls (without previous ischemic cerebrovascular disease) underwent 3T MRI to visualize atherosclerotic intracranial vessel wall lesions (VWLs). We compared the presence and burden of VWLs (total and for the anterior cerebral, middle cerebral, intracranial internal carotid, posterior cerebral, and basilar arteries separately) between PXE patients and healthy controls using logistic (presence versus absence) and negative binomial regression models (VWL count) adjusted for relevant confounders. All regressions included group (PXE patients vs. healthy controls) as independent variable.
RESULTS: We found that 34 (68.0%) PXE patients and 28 (70.0%) healthy controls had a VWL (odds ratio for presence 1.06 [95%CI 0.38-2.91]). In addition, the total burden of VWLs was similar between PXE patients (68 VWLs) and healthy controls (73 VWLs, incidence rate ratio for count 0.81 [95%CI 0.55-1.20]). Findings were similar when analyses were stratified for artery.
CONCLUSIONS: The distribution and burden of intracranial atherosclerosis were similar between PXE patients and healthy controls. This implies PXE and its underlying mutations do not involve increased (intracranial) atherogenesis and that vascular calcification or other mechanisms explains the increased stroke risk in PXE.
| Medienart: |
E-Artikel |
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| Erscheinungsjahr: |
2022 |
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| Erschienen: |
2022 |
| Enthalten in: |
Zur Gesamtaufnahme - volume:350 |
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| Enthalten in: |
Atherosclerosis - 350(2022) vom: 05. Juni, Seite 19-24 |
| Sprache: |
Englisch |
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| Beteiligte Personen: |
Lucci, Carlo [VerfasserIn] |
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| Links: |
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| Themen: |
Intracranial atherosclerosis |
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| Anmerkungen: |
Date Completed 03.06.2022 Date Revised 22.06.2022 published: Print-Electronic Citation Status MEDLINE |
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| doi: |
10.1016/j.atherosclerosis.2022.04.014 |
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| funding: |
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| Förderinstitution / Projekttitel: |
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| PPN (Katalog-ID): |
NLM339975938 |
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| 100 | 1 | |a Lucci, Carlo |e verfasserin |4 aut | |
| 245 | 1 | 0 | |a Intracranial atherosclerosis in pseudoxanthoma elasticum |b A case-control study |
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| 500 | |a published: Print-Electronic | ||
| 500 | |a Citation Status MEDLINE | ||
| 520 | |a Copyright © 2022 The Authors. Published by Elsevier B.V. All rights reserved. | ||
| 520 | |a BACKGROUND AND AIMS: Pseudoxanthoma elasticum (PXE) is a genetic disorder characterized by systemic calcification of elastin fibers. Additionally, PXE is associated with an increased risk of stroke. It has been hypothesized that this may be caused by accelerated (intracranial) atherogenesis, as a consequence of specific genetic mutations underlying PXE. Hence, we compared the distribution and burden of intracranial atherosclerosis between PXE patients and healthy controls | ||
| 520 | |a METHODS: Fifty PXE patients and 40 age-and-sex-matched healthy controls (without previous ischemic cerebrovascular disease) underwent 3T MRI to visualize atherosclerotic intracranial vessel wall lesions (VWLs). We compared the presence and burden of VWLs (total and for the anterior cerebral, middle cerebral, intracranial internal carotid, posterior cerebral, and basilar arteries separately) between PXE patients and healthy controls using logistic (presence versus absence) and negative binomial regression models (VWL count) adjusted for relevant confounders. All regressions included group (PXE patients vs. healthy controls) as independent variable | ||
| 520 | |a RESULTS: We found that 34 (68.0%) PXE patients and 28 (70.0%) healthy controls had a VWL (odds ratio for presence 1.06 [95%CI 0.38-2.91]). In addition, the total burden of VWLs was similar between PXE patients (68 VWLs) and healthy controls (73 VWLs, incidence rate ratio for count 0.81 [95%CI 0.55-1.20]). Findings were similar when analyses were stratified for artery | ||
| 520 | |a CONCLUSIONS: The distribution and burden of intracranial atherosclerosis were similar between PXE patients and healthy controls. This implies PXE and its underlying mutations do not involve increased (intracranial) atherogenesis and that vascular calcification or other mechanisms explains the increased stroke risk in PXE | ||
| 650 | 4 | |a Journal Article | |
| 650 | 4 | |a Intracranial atherosclerosis | |
| 650 | 4 | |a MRI | |
| 650 | 4 | |a Pseudoxanthoma elasticum | |
| 650 | 4 | |a Stroke | |
| 650 | 4 | |a Vessel wall lesions | |
| 700 | 1 | |a van den Beukel, Tim C |e verfasserin |4 aut | |
| 700 | 1 | |a Bartstra, Jonas W |e verfasserin |4 aut | |
| 700 | 1 | |a Zwanenburg, Jaco |e verfasserin |4 aut | |
| 700 | 1 | |a van der Kolk, Anja |e verfasserin |4 aut | |
| 700 | 1 | |a Takx, Richard |e verfasserin |4 aut | |
| 700 | 1 | |a Hendrikse, Jeroen |e verfasserin |4 aut | |
| 700 | 1 | |a Geerlings, Mirjam I |e verfasserin |4 aut | |
| 700 | 1 | |a Bos, Daniel |e verfasserin |4 aut | |
| 700 | 1 | |a Spiering, Wilko |e verfasserin |4 aut | |
| 700 | 1 | |a de Jong, Pim A |e verfasserin |4 aut | |
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