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August 29, 2023; 101 (9) Research Article

Aneurysm Prevalence and Quality of Life During Screening in Relatives of Patients With Unruptured Intracranial Aneurysms

A Prospective Study

Liselore A. Mensing, Rick J. van Tuijl, View ORCID ProfileJacoba P. Greving, Birgitta K. Velthuis, View ORCID ProfileIrene C. van der Schaaf, View ORCID ProfileMarieke J.H. Wermer, View ORCID ProfileDagmar Verbaan, View ORCID ProfileW. Peter Vandertop, Nicolaas P.A. Zuithoff, View ORCID ProfileGabriël J.E. Rinkel, Ynte M. Ruigrok
First published July 6, 2023, DOI: https://doi.org/10.1212/WNL.0000000000207475
Liselore A. Mensing
From the UMC Utrecht Brain Center (L.A.M., G.J.E.R., Y.M.R.), Department of Neurology and Neurosurgery, Department of Radiology (R.J.v.T., B.K.V., I.C.v.d.S.), and Julius Center for Health Sciences and Primary Care (J.P.G., N.P.A.Z.), University Medical Center Utrecht; Department of Neurology (M.J.H.W.), University Medical Center Leiden; and Department of Neurosurgery (D.V., W.P.V.), Amsterdam University Medical Center, the Netherlands.
MD
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Rick J. van Tuijl
From the UMC Utrecht Brain Center (L.A.M., G.J.E.R., Y.M.R.), Department of Neurology and Neurosurgery, Department of Radiology (R.J.v.T., B.K.V., I.C.v.d.S.), and Julius Center for Health Sciences and Primary Care (J.P.G., N.P.A.Z.), University Medical Center Utrecht; Department of Neurology (M.J.H.W.), University Medical Center Leiden; and Department of Neurosurgery (D.V., W.P.V.), Amsterdam University Medical Center, the Netherlands.
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Jacoba P. Greving
From the UMC Utrecht Brain Center (L.A.M., G.J.E.R., Y.M.R.), Department of Neurology and Neurosurgery, Department of Radiology (R.J.v.T., B.K.V., I.C.v.d.S.), and Julius Center for Health Sciences and Primary Care (J.P.G., N.P.A.Z.), University Medical Center Utrecht; Department of Neurology (M.J.H.W.), University Medical Center Leiden; and Department of Neurosurgery (D.V., W.P.V.), Amsterdam University Medical Center, the Netherlands.
PhD
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  • ORCID record for Jacoba P. Greving
Birgitta K. Velthuis
From the UMC Utrecht Brain Center (L.A.M., G.J.E.R., Y.M.R.), Department of Neurology and Neurosurgery, Department of Radiology (R.J.v.T., B.K.V., I.C.v.d.S.), and Julius Center for Health Sciences and Primary Care (J.P.G., N.P.A.Z.), University Medical Center Utrecht; Department of Neurology (M.J.H.W.), University Medical Center Leiden; and Department of Neurosurgery (D.V., W.P.V.), Amsterdam University Medical Center, the Netherlands.
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Irene C. van der Schaaf
From the UMC Utrecht Brain Center (L.A.M., G.J.E.R., Y.M.R.), Department of Neurology and Neurosurgery, Department of Radiology (R.J.v.T., B.K.V., I.C.v.d.S.), and Julius Center for Health Sciences and Primary Care (J.P.G., N.P.A.Z.), University Medical Center Utrecht; Department of Neurology (M.J.H.W.), University Medical Center Leiden; and Department of Neurosurgery (D.V., W.P.V.), Amsterdam University Medical Center, the Netherlands.
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Marieke J.H. Wermer
From the UMC Utrecht Brain Center (L.A.M., G.J.E.R., Y.M.R.), Department of Neurology and Neurosurgery, Department of Radiology (R.J.v.T., B.K.V., I.C.v.d.S.), and Julius Center for Health Sciences and Primary Care (J.P.G., N.P.A.Z.), University Medical Center Utrecht; Department of Neurology (M.J.H.W.), University Medical Center Leiden; and Department of Neurosurgery (D.V., W.P.V.), Amsterdam University Medical Center, the Netherlands.
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Dagmar Verbaan
From the UMC Utrecht Brain Center (L.A.M., G.J.E.R., Y.M.R.), Department of Neurology and Neurosurgery, Department of Radiology (R.J.v.T., B.K.V., I.C.v.d.S.), and Julius Center for Health Sciences and Primary Care (J.P.G., N.P.A.Z.), University Medical Center Utrecht; Department of Neurology (M.J.H.W.), University Medical Center Leiden; and Department of Neurosurgery (D.V., W.P.V.), Amsterdam University Medical Center, the Netherlands.
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W. Peter Vandertop
From the UMC Utrecht Brain Center (L.A.M., G.J.E.R., Y.M.R.), Department of Neurology and Neurosurgery, Department of Radiology (R.J.v.T., B.K.V., I.C.v.d.S.), and Julius Center for Health Sciences and Primary Care (J.P.G., N.P.A.Z.), University Medical Center Utrecht; Department of Neurology (M.J.H.W.), University Medical Center Leiden; and Department of Neurosurgery (D.V., W.P.V.), Amsterdam University Medical Center, the Netherlands.
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Nicolaas P.A. Zuithoff
From the UMC Utrecht Brain Center (L.A.M., G.J.E.R., Y.M.R.), Department of Neurology and Neurosurgery, Department of Radiology (R.J.v.T., B.K.V., I.C.v.d.S.), and Julius Center for Health Sciences and Primary Care (J.P.G., N.P.A.Z.), University Medical Center Utrecht; Department of Neurology (M.J.H.W.), University Medical Center Leiden; and Department of Neurosurgery (D.V., W.P.V.), Amsterdam University Medical Center, the Netherlands.
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Gabriël J.E. Rinkel
From the UMC Utrecht Brain Center (L.A.M., G.J.E.R., Y.M.R.), Department of Neurology and Neurosurgery, Department of Radiology (R.J.v.T., B.K.V., I.C.v.d.S.), and Julius Center for Health Sciences and Primary Care (J.P.G., N.P.A.Z.), University Medical Center Utrecht; Department of Neurology (M.J.H.W.), University Medical Center Leiden; and Department of Neurosurgery (D.V., W.P.V.), Amsterdam University Medical Center, the Netherlands.
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Ynte M. Ruigrok
From the UMC Utrecht Brain Center (L.A.M., G.J.E.R., Y.M.R.), Department of Neurology and Neurosurgery, Department of Radiology (R.J.v.T., B.K.V., I.C.v.d.S.), and Julius Center for Health Sciences and Primary Care (J.P.G., N.P.A.Z.), University Medical Center Utrecht; Department of Neurology (M.J.H.W.), University Medical Center Leiden; and Department of Neurosurgery (D.V., W.P.V.), Amsterdam University Medical Center, the Netherlands.
MD, PhD
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Citation
Aneurysm Prevalence and Quality of Life During Screening in Relatives of Patients With Unruptured Intracranial Aneurysms
A Prospective Study
Liselore A. Mensing, Rick J. van Tuijl, Jacoba P. Greving, Birgitta K. Velthuis, Irene C. van der Schaaf, Marieke J.H. Wermer, Dagmar Verbaan, W. Peter Vandertop, Nicolaas P.A. Zuithoff, Gabriël J.E. Rinkel, Ynte M. Ruigrok
Neurology Aug 2023, 101 (9) e904-e912; DOI: 10.1212/WNL.0000000000207475

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Abstract

Background and Objectives Screening for unruptured intracranial aneurysms (UIAs) is effective for first-degree relatives (FDRs) of patients with aneurysmal subarachnoid hemorrhage (aSAH). Whether screening is also effective for FDRs of patients with UIA is unknown. We determined the yield of screening in such FDRs, assessed rupture risk and treatment decisions of aneurysms that were found, identified potential high-risk subgroups, and studied the effects of screening on quality of life (QoL).

Methods In this prospective cohort study, we included FDRs, aged 20–70 years, of patients with UIA without a family history of aSAH who visited the Neurology outpatient clinic in 1 of 3 participating tertiary referral centers in the Netherlands. FDRs were screened for UIA with magnetic resonance angiography between 2017 and 2021. We determined UIA prevalence and developed a prediction model for UIA risk at screening using multivariable logistic regression. QoL was evaluated with questionnaires 6 times during the first year after screening and assessed with a linear mixed-effects model.

Results We detected 24 UIAs in 23 of 461 screened FDRs, resulting in a 5.0% prevalence (95% CI 3.2–7.4). The median aneurysm size was 3 mm (interquartile range [IQR] 2–4 mm), and the median 5-year rupture risk assessed with the PHASES score was 0.7% (IQR 0.4%–0.9%). All UIAs received follow-up imaging, and none were treated preventively. After a median follow-up of 24 months (IQR 13–38 months), no UIA had changed. Predicted UIA risk at screening ranged between 2.3% and 14.7% with the highest risk in FDRs who smoke and have excessive alcohol consumption (c-statistic: 0.76; 95% CI 0.65–0.88). At all survey moments, health-related QoL and emotional functioning were comparable with those in a reference group from the general population. One FDR with a positive screening result expressed regret about screening.

Discussion Based on the current data, we do not advise screening FDRs of patients with UIA because all identified UIAs had a low rupture risk. We observed no negative effect of screening on QoL. A longer follow-up should determine the risk of aneurysm growth requiring preventive treatment.

Glossary

aSAH=
aneurysmal subarachnoid hemorrhage;
COVID-19=
coronavirus disease 2019;
EQ-5D=
EuroQoL 5 Dimensions;
EQ-VAS=
EuroQoL Visual Analog Scale;
HADS=
Hospital Anxiety and Depression Scale;
FDR=
first-degree relative;
HRQoL=
health-related QoL;
IQR=
interquartile range;
LME=
linear mixed-effect model;
MRA=
magnetic resonance angiography;
PCKD=
polycystic kidney disease;
QoL=
quality of life;
UIA=
unruptured intracranial aneurysm;
UMCU=
University Medical Center Utrecht;
USER-P=
Utrecht Scale for Evaluation of Rehabilitation—Participation

Footnotes

  • Go to Neurology.org/N for full disclosures. Funding information and disclosures deemed relevant by the authors, if any, are provided at the end of the article.

  • Submitted and externally peer reviewed. The handling editor was Editor-in-Chief José Merino, MD, MPhil, FAAN.

  • Patient Page page e986

  • Received October 28, 2022.
  • Accepted in final form April 17, 2023.
  • © 2023 American Academy of Neurology
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