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烟雾病伴颅内动脉瘤的临床特点、预后危险因素及再出血生存分析
Clinical Characteristics,Prognostic Risk Factors and Survival Analysis of Rebleeding in Moyamoya Disease with Intracranial Aneurysms
【作者】 姚远;
【导师】 王林;
【作者基本信息】 浙江大学 , 临床医学(专业学位), 2020, 硕士
【摘要】 背景与目的据统计约3.4%-14.8%的烟雾病患者伴有颅内动脉瘤,该亚型具有较高的颅内出血倾向,且术后易发生再出血。如何处理动脉瘤,减少患者的再出血风险,是目前亟需攻克的难题。本研究分析了烟雾病伴动脉瘤的临床特征,探究影响预后及再出血的独立危险因素,并通过对手术方案的探讨,为临床治疗决策提供依据。方法回顾性研究2011年03月31日至2019年3月31日于浙江大学医学院附属第二医院神经外科入院治疗的337例烟雾病病例,排除未诊断伴动脉瘤286例、资料不全9例、失访1例,最后纳入符合要求的患者41例。描述分析其临床特征,并采用单因素及多因素分析法,筛选影响患者预后及再出血的独立危险因素。后采用Kaplan-Meier法行再出血生存分析,用log-rank法比较不同分组间生存曲线的差异,再将有意义的因素纳入Cox比例风险模型行再出血多因素分析,从而探究影响烟雾病伴动脉瘤再出血风险的独立危险因素,最后通过典型病例探讨手术方案的抉择。结果41例烟雾病伴动脉瘤患者中出血型27例,缺血型14例。平均发病年龄为49.1±10.2岁,男女比例0.64:1。入院格拉斯哥昏迷指数评分中位数为13分,烟雾病改良Suzuki分期中位分期为3期。41例患者中动脉瘤位于主干动脉的有30例,位于外周动脉4例,7例为多发动脉瘤,其中动脉瘤破裂13例。手术治疗31例,保守治疗10例。手术治疗中仅行搭桥15例,搭桥联合动脉瘤开颅夹闭9例,3例仅动脉瘤开颅夹闭,3例行搭桥联合动脉瘤介入栓塞,1例仅动脉瘤介入栓塞。共23例发生并发症,短期并发症10例,远期并发症18例。其中再出血12例,死亡6例。入院GCS评分(P=0.023)为短期并发症的独立危险因素。影响患者远期神经功能改善情况的独立危险因素是再出血(P=0.003)。最后,我们发现烟雾状血管分布范围(OR=23.703,95CI%(1.413~397.740),P=0.028)较大的患者再出血风险高,而高入院GCS评分(OR=15.430,95CI%(1.377~172.900),P=0.026)的患者再出血风险往往较低。受限于病例量少,本研究未能得出手术方式与患者预后及再出血有统计学相关性的结论,因此我们以典型病例阐述我院单中心治疗经验。结论本文记录并描述了烟雾病伴动脉瘤患者的临床特征,并发现入院GCS评分与短期并发症的发生率相关,再出血可显著影响患者预后神经功能。我们还发现入院GCS评分及烟雾状血管分布范围与患者再出血有相关,既往出血史为患者再出血的潜在危险因素。烟雾病伴动脉瘤的治疗方式有多种,应该根据不同患者情况选择最佳的治疗方案。
【Abstract】 Objective According to statistics,about 3.4%-14.8% of patients with moyamoya disease are associated with intracranial aneurysms.This subtype has a higher tendency for intracranial hemorrhage and is prone to rebleeding after surgery.How to deal with aneurysms and reduce the risk of rebleeding in patients is a difficult problem to be overcome.This study described the clinical characteristics of moyamoya disease with aneurysms,explored independent risk factors that affect prognosis and rebleeding,and provided a basis for clinical treatment decisions through the discussion of surgical options.Methods A retrospective study of 337 cases of moyamoya disease admitted to the Department of Neurosurgery of The Second Affiliated Hospital of Zhejiang University School of Medicine from March 31,2011 to March 31,2019,286 cases with undiagnosed aneurysms,9 cases with incomplete data,1 case lost to follow-up,and finally 41 eligible patients were included.Describe and analyze its clinical characteristics,and use single-factor and multi-factor analysis to screen independent risk factors that affect patients’ prognosis and rebleeding.Kaplan-Meier method was then used to perform rebleeding survival analysis,log-rank method was used to compare the differences in survival curves between different groups,and meaningful factors were incorporated into the Cox proportional hazard model for rebleeding multivariate analysis to explore the effects of moyamoya disease partners.Independent risk factors for aneurysm rebleeding risk.Finally,the choice of surgical scheme was explored through typical cases.Results Of the 41 patients with moyamoya disease and aneurysm,27 were hemorrhagic and 14 were ischemic.The mean age of onset was 49.1 ± 10.2 years,and the male to female ratio was 0.64:1.The median Glasgow coma index score was 13 points,and the median staging of the mozambique-modified Suzuki stage was 3 stages.Of the 41 patients,30 had aneurysms in the main artery,4 had peripheral arteries,and 7 had multiple aneurysms,of which 13 had aneurysm rupture.Surgical treatment was performed in 31 cases and conservative treatment in 10 cases.Only 15 cases of bypass grafting were performed during surgery,9 cases of bypass graft and aneurysm craniotomy,3 cases of aneurysm craniotomy,3 cases of bypass and aneurysm interventional embolization,and 1 case of aneurysm interventional embolization.Complications occurred in 23 cases,short-term complications in 10 cases,and long-term complications in 18 cases.Of these,12 patients had rebleeding and 6 died.The admission GCS score(P = 0.023)was an independent risk factor for short-term complications.An independent risk factor for patients with long-term neurologic improvement was rebleeding(P = 0.003).Finally,we found that patients with larger smoke-like blood vessel distribution range(OR = 23.703,95CI%(1.413 ~ 397.740),P =0.028)had a higher risk of rebleeding,and a high admission GCS score(OR = 15.430,95CI%(1.377 ~ 172.900),P = 0.026).Limited by the small number of cases,this study could not draw a conclusion that the surgical method has statistical correlation with the patient’s prognosis and rebleeding.Therefore,we described our hospital’s single-center treatment experience with typical cases.Conclusions This article recorded and described the clinical characteristics of patients with moyamoya disease and aneurysms,and found that the admission GCS score is related to the incidence of short-term complications,and rebleeding can significantly affect the prognosis of neurological function.We also found that the admission GCS score and the range of smoke-like blood vessels were related to the patient’s rebleeding.Previous bleeding history was a potential risk factor for patients’ rebleeding.There are many treatments for moyamoya disease with aneurysms,and the best treatment plan should be selected according to the situation of different patients.
【Key words】 Moyamoya Disease; Intracranial Aneurysm; Rebleeding; Risk Factors; Survival Analysis;