Untreated clinical course of cerebral cavernous malformations: a prospective, population-based cohort study.
Untreated clinical course of cerebral cavernous malformations: a prospective, population-based cohort study.
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DOI：
10.1016/s1474-4422(12)70004-2
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发表时间：
2012-03
期刊：
影响因子：
48
通讯作者：
Warlow, Charles P.
中科院分区：
文献类型：
--
作者：
Salman, Rustam Al-Shahi;Hall, Julie M.;Horne, Margaret A.;Moultrie, Fiona;Josephson, Colin B.;Bhattacharya, Joj;Counsell, Carl E.;Murray, Gordon D.;Papanastassiou, Vakis;Ritchie, Vaughn;Roberts, Richard C.;Sellar, Robin J.;Warlow, Charles P.
脑海绵状畸形（CCM）易于出血，但颅内出血和局灶性神经功能缺损的风险以及可能预测其发生的因素尚不清楚。我们的目的是量化这些风险，并调查它们是否受性别和CCM位置的影响。我们进行了一项基于人群的研究，使用多个重叠的病例确定来源（包括苏格兰范围内的神经学家，神经外科医生，中风医生，放射科医生和病理学家的合作，以及医院出院和死亡证明的登记册的搜索），以确定明确的CCM诊断首先在1999年和2003年之间在苏格兰居民，研究神经放射科医生独立验证。我们使用了多个来源的前瞻性随访，以确定结果事件（通过使用脑成像，由研究人员对潜在的预测因素进行评估）并评估成人的依赖性。主要结局是与CCM明确或可能相关的颅内出血或局灶性神经功能缺损（不包括癫痫发作）的复合终点。139名成人至少有一个明确的CCM，134人在最初的表现还活着。在1177人-年的随访中（完整性97%），仅颅内出血的5年首次出血风险低于复发出血风险（2.4%，95% CI 0.0 - 5.7 vs 29.5%，4.1 - 55.0; p<0.0001）。对于主要结局，首次事件的5年风险低于复发风险（9.3%，3.1 - 15.4 vs 42.4%，26.8 - 58.0; p<0.0001）。主要结局的年复发风险从第1年的19.8%（95%CI 6.1 - 33.4）下降至第5年的5.0%（0.0 - 14.8），女性高于男性（p= 0.01），但脑干CCM成人与其他部位的CCM相比无差异（p= 0.17）。CCM引起的复发性颅内出血或局灶性神经功能缺损的风险大于首次事件的风险，女性高于男性，并在5年内下降。这些信息可以用于临床实践，但需要进一步的工作来精确量化长期风险，并了解为什么女性比男性更容易复发。英国医学研究理事会、苏格兰政府首席科学家办公室和英国中风协会。
Cerebral cavernous malformations (CCMs) are prone to bleeding but the risk of intracranial haemorrhage and focal neurological deficits, and the factors that might predict their occurrence, are unclear. We aimed to quantify these risks and investigate whether they are affected by sex and CCM location. We undertook a population-based study using multiple overlapping sources of case ascertainment (including a Scotland-wide collaboration of neurologists, neurosurgeons, stroke physicians, radiologists, and pathologists, as well as searches of registers of hospital discharges and death certificates) to identify definite CCM diagnoses first made in Scottish residents between 1999 and 2003, which study neuroradiologists independently validated. We used multiple sources of prospective follow-up both to identify outcome events (which were assessed by use of brain imaging, by investigators masked to potential predictive factors) and to assess adults' dependence. The primary outcome was a composite of intracranial haemorrhage or focal neurological deficits (not including epileptic seizure) that were definitely or possibly related to CCM. 139 adults had at least one definite CCM and 134 were alive at initial presentation. During 1177 person-years of follow-up (completeness 97%), for intracranial haemorrhage alone the 5-year risk of a first haemorrhage was lower than the risk of recurrent haemorrhage (2·4%, 95% CI 0·0–5·7 vs 29·5%, 4·1–55·0; p<0·0001). For the primary outcome, the 5-year risk of a first event was lower than the risk of recurrence (9·3%, 3·1–15·4 vs 42·4%, 26·8–58·0; p<0·0001). The annual risk of recurrence of the primary outcome declined from 19·8% (95% CI 6·1–33·4) in year 1 to 5·0% (0·0–14·8) in year 5 and was higher for women than men (p=0·01) but not for adults with brainstem CCMs versus CCMs in other locations (p=0·17). The risk of recurrent intracranial haemorrhage or focal neurological deficit from a CCM is greater than the risk of a first event, is greater for women than for men, and declines over 5 years. This information can be used in clinical practice, but further work is needed to quantify risks precisely in the long term and to understand why women are at greater risk of recurrence than men. UK Medical Research Council, Chief Scientist Office of the Scottish Government, and UK Stroke Association.