UMC Utrecht Brain Center, Department of Neurology and Neurosurgery, University Medical Center Utrecht, the Netherlands (C.C.M.Z., G.J.E.R., Y.M.R.).
Department of Diagnostic and Therapeutic Neuroradiology, University Hospital of Nantes, L'institut du thorax, Nantes, Pays de la Loire, FR (R.B., P.C.D.B., R.R., H.D.).
Stroke. 2022 May;53(5):1645-1650. doi: 10.1161/STROKEAHA.121.034612. Epub 2022 Feb 11.
Persons with a positive family history of aneurysmal subarachnoid hemorrhage are at increased risk of aneurysmal subarachnoid hemorrhage. Preventive screening for intracranial aneurysms (IAs) in these persons is cost-effective but not very efficient. We aimed to develop and externally validate a model for predicting the probability of an IA at first screening in persons with a positive family history of aneurysmal subarachnoid hemorrhage.
For model development, we studied results from initial screening for IA in 660 prospectively collected persons with ≥2 affected first-degree relatives screened at the University Medical Center Utrecht. For validation, we studied results from 258 prospectively collected persons screened in the University Hospital of Nantes. We assessed potential predictors of IA presence in multivariable logistic regression analysis. Predictive performance was assessed with the C statistic and a calibration plot and corrected for overfitting.
IA were present in 79 (12%) persons in the development cohort. Predictors were number of affected relatives, age, smoking, and hypertension (NASH). The NASH score had a C statistic of 0.68 (95% CI, 0.62-0.74) and showed good calibration in the development data. Predicted probabilities of an IA at first screening varied from 5% in persons aged 20 to 30 years with two affected relatives, without hypertension who never smoked, up to 36% in persons aged 60 to 70 years with ≥3 affected relatives, who have hypertension and smoke(d). In the external validation data IA were present in 67 (26%) persons, the model had a C statistic of 0.64 (95% CI, 0.57-0.71) and slightly underestimated IAs risk.
For persons with ≥2 affected first-degree relatives, the NASH score improves current predictions and provides risk estimates for an IA at first screening between 5% and 36% based on 4 easily retrievable predictors. With the information such persons can now make a better informed decision about whether or not to undergo preventive screening.
有蛛网膜下腔出血阳性家族史的人发生蛛网膜下腔出血的风险增加。对这些人进行颅内动脉瘤(IA)预防性筛查具有成本效益,但效率不高。我们旨在开发和外部验证一种用于预测有蛛网膜下腔出血阳性家族史的人首次筛查时 IA 概率的模型。
为了进行模型开发,我们研究了在乌得勒支大学医学中心前瞻性收集的 660 名至少有 2 名受影响一级亲属接受筛查的患者中,首次筛查 IA 的结果。为了验证,我们研究了在南特大学医院前瞻性收集的 258 名接受筛查的患者的结果。我们在多变量逻辑回归分析中评估了 IA 存在的潜在预测因素。使用 C 统计量和校准图评估预测性能,并进行过度拟合校正。
在发展队列中,79 名(12%)患者存在 IA。预测因素包括受影响亲属的数量、年龄、吸烟和高血压(NASH)。NASH 评分的 C 统计量为 0.68(95%CI,0.62-0.74),并且在发展数据中具有良好的校准。首次筛查时 IA 的预测概率从 20 至 30 岁、无高血压且从不吸烟、有 2 名受影响亲属的患者的 5%,到 60 至 70 岁、有≥3 名受影响亲属、有高血压且吸烟的患者的 36%不等。在外部验证数据中,67 名(26%)患者存在 IA,该模型的 C 统计量为 0.64(95%CI,0.57-0.71),并且略微低估了 IA 的风险。
对于有≥2 名受影响一级亲属的患者,NASH 评分可改善当前的预测结果,并根据 4 个易于获取的预测因素提供首次筛查时 IA 概率在 5%至 36%之间的风险估计。有了这些信息,患者现在可以更好地决定是否进行预防性筛查。