Patient and aneurysm characteristics in familial intracranial aneurysms. A systematic review and meta-analysis

Background and purposePatient and aneurysm characteristics have been reported to differ between patients with familial and non-familial intracranial aneurysms (IAs), although results are inconsistent. We systematically reviewed and meta-analyzed the literature to identify and quantify patient- and aneurysm characteristics associated with familial IAs.MethodsWe searched PubMed and EMBASE for case-control and cohort studies comparing patient- and aneurysm characteristics between familial and non-familial IAs. Two observers independently assessed study eligibility and appraised quality with the Newcastle Ottawa Scale. With univariable weighted linear regression analysis we calculated β-coefficients with corresponding 95% confidence intervals (CIs) for ruptured and unruptured IAs combined and for ruptured IAs only. Heterogeneity was assessed with Higgins I2.ResultsA total of 15 articles were included in the meta-analysis in which 16,346 patients were analyzed with a total of 14,225 IAs. For ruptured and unruptured IAs combined, multiple IAs were more prevalent in familial (28.5%) than in non-familial IAs (20.4%; β = 0.10, 95% CI, 0.04 to 0.16; I2 0%). For ruptured IAs only, in familial patients IAs were more prevalent on the middle cerebral artery (41.1% versus 29.5%; β = 0.12, 95% CI, 0.01 to 0.24; I2 12%) and ruptured at a younger age (46.5 years versus 50.8 years; β = -5.00, 95% CI, -9.31 to -0.69; I2 98%) than in non-familial patients. No significant differences were found for the proportion of women, size of the aneurysm at time of rupture, smoking or hypertension.ConclusionThese results suggest that characteristics of familial and non-familial IAs show considerable overlap, yet differ on specific aspects. However, results for age at rupture showed considerable heterogeneity. These findings should be taken into consideration for future etiological research into IAs.

背景与目的：已有研究报道家族性与非家族性颅内动脉瘤（intracranial aneurysms, IAs）患者的临床特征及动脉瘤特征存在差异，但相关研究结果并不一致。本研究对现有文献进行系统综述与荟萃分析，旨在明确并量化与家族性颅内动脉瘤相关的患者及动脉瘤特征。
方法：我们在PubMed和EMBASE数据库中检索对比家族性与非家族性颅内动脉瘤患者及动脉瘤特征的病例对照研究与队列研究。由两名研究者独立评估研究的纳入资格，并采用纽卡斯尔-渥太华量表（Newcastle Ottawa Scale）对研究质量进行评价。我们采用单变量加权线性回归分析，分别针对合并的破裂与未破裂颅内动脉瘤，以及仅破裂颅内动脉瘤两种情况，计算β系数及对应的95%置信区间（confidence intervals, CIs）。采用Higgins I2统计量评估研究间异质性。
结果：本荟萃分析共纳入15篇文献，涉及16346例患者，共计14225个颅内动脉瘤。在合并的破裂与未破裂颅内动脉瘤中，家族性颅内动脉瘤患者的多发动脉瘤患病率（28.5%）高于非家族性患者（20.4%；β=0.10，95%CI：0.04~0.16；I²=0%）。仅针对破裂颅内动脉瘤的分析显示，家族性患者的动脉瘤更常发生于大脑中动脉（41.1% vs 29.5%；β=0.12，95%CI：0.01~0.24；I²=12%），且动脉瘤破裂时患者年龄更小（46.5岁 vs 50.8岁；β=-5.00，95%CI：-9.31~-0.69；I²=98%），相较于非家族性患者。在女性占比、破裂时动脉瘤大小、吸烟史或高血压病史方面未发现显著差异。
结论：本研究结果表明，家族性与非家族性颅内动脉瘤的特征存在较大重叠，但在特定方面存在差异。不过，动脉瘤破裂年龄的相关结果存在显著异质性。上述发现可为未来颅内动脉瘤的病因学研究提供参考。