Open AccessHeritability of territory of ruptured and unruptured intracranial aneurysms in families
Author(s) -
Mayte Sánchez van Kammen,
Romain Bourcier,
Charles J. Moomaw,
Joseph P. Broderick,
Daniel Woo,
Chrysanthi Papagiannaki,
O. Lévrier,
Antti Lindgren,
Timo Koivisto,
Juha E. Jääskeläinen,
Gabriël J.E. Rinkel,
Ynte M. Ruigrok
Publication year - 2020
Publication title -
plos one
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 0.99
H-Index - 332
ISSN - 1932-6203
DOI - 10.1371/journal.pone.0236714
Subject(s) - aneurysm , concordance , medicine , odds ratio , subarachnoid hemorrhage , surgery
Background A previous study suggested that intracranial aneurysms are more likely to occur in the same arterial territory within families. We aimed to replicate this analysis in independent families and in a sample limited to intracranial aneurysms that ruptured. Methods Among families with ≥2 first-degree relatives with intracranial aneurysms, we randomly matched index families to comparison families, and compared concordance in intracranial aneurysm territory between index and comparison families using a conditional logistic events/trials model. We analyzed three European cohorts separately, and pooled the results with those of the Familial Intracranial Aneurysm study by performing an inverse variance fixed effects meta-analysis. The main analysis included both unruptured and ruptured intracranial aneurysms, and a secondary analysis only ruptured intracranial aneurysms. Results Among 70 Dutch, 142 Finnish, and 34 French families, concordance regarding intracranial aneurysm territory was higher within families than between families, although not statistically significant. Meta-analysis revealed higher concordance in territory within families overall (odds ratio [OR] 1.7, 95%CI 1.3–2.2) and for each separate territory except the anterior cerebral artery. In the analysis of ruptured intracranial aneurysms, overall territory concordance was higher within families than between families (OR 1.8; 95%CI 1.1–2.7) but the territory-specific analysis showed statistical significance only for the internal carotid artery territory. Conclusions We confirmed that familial intracranial aneurysms are more likely to occur in the same arterial territory within families. Moreover, we found that ruptured aneurysms were also more likely to occur in the same arterial territory within families.