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首页> 外文期刊>European journal of neurology: the official journal of the European Federation of Neurological Societies >Neurosarcoidosis-related intracranial haemorrhage: Three new cases and a systematic review of the literature
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Neurosarcoidosis-related intracranial haemorrhage: Three new cases and a systematic review of the literature

机译：神经结节病相关的颅内出血：三例新病例并文献复习

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Background and purpose: Intracranial haemorrhage in neurosarcoidosis (NS-ICH) is rare, poorly understood and the diagnosis of NS may not be immediately apparent. Methods: The clinical features of three new NS-ICH cases are described including new neuropathological findings and collated with cases from a systematic literature review. Results: Cases: (i) A 41-year-old man with headaches, hypoandrogenism and encephalopathy developed a cerebellar haemorrhage. He had neuropathological confirmation of NS with biopsy-proven angiocentric granulomata and venous disruption. He responded to immunosuppressive therapy. (ii) A 41-year-old man with no history of hypertension was found unconscious. A subsequently fatal pontine haemorrhage was diagnosed. Liver biopsy revealed sarcoid granulomas. (iii) A 36-year-old man with raised intracranial pressure headaches presented with a seizure and a frontal haemorrhage. Hilar lymph node biopsy confirmed sarcoidosis, and he was treated successfully. Systematic review: Twelve other published cases were identified and collated with our cases. Average age was 36 years and M:F = 2.3:1; 46% presented with neurological symptoms and 31% had CNS-isolated disease. Immediate symptoms of ICH were acute/worsening headache or seizures (60%). ICH was supratentorial (62%), infratentorial (31%) or subarachnoid (7%). Forty percent had definite NS, 53% probable NS and 7% possible NS (Zajicek criteria). Antigranulomatous/immunosuppressive therapy regimens varied and 31% died. Conclusions: This series expands our knowledge of the pathology of NS-ICH, which may be of arterial or venous origin. One-third have isolated NS. Clinicians should consider NS in young-onset ICH because early aggressive antigranulomatous therapy may improve outcome.
机译：背景与目的：神经结节病（NS-ICH）的颅内出血很少见，了解甚少，对NS的诊断可能不会立即显现。方法：描述了3例新的NS-ICH患者的临床特征，包括新的神经病理学发现，并与来自系统文献回顾的病例进行对照。结果：病例：（i）一名头痛，低雄激素血症和脑病的41岁男子发生了小脑出血。他通过活检证实的血管中心性肉芽肿和静脉破裂得到了神经病理学证实的NS。他对免疫抑制疗法有反应。 （ii）一名无高血压病史的41岁男子被发现昏迷。诊断出随后致命的桥脑出血。肝活检显示结节性肉芽肿。 （iii）一名36岁的男子，颅内压升高，表现为癫痫发作和额叶出血。肺门淋巴结活检证实结节病，他得到了成功治疗。系统评价：确定了其他十二个已发布案例，并与我们的案例进行比较。平均年龄为36岁，男：女= 2.3：1;有46％的患者出现神经系统症状，而31％的患者患有中枢神经系统隔离疾病。 ICH的直接症状是急性/加重性头痛或癫痫发作（60％）。 ICH为幕上性（62％），下腹式（31％）或蛛网膜下腔（7％）。 40％的患者具有确定的NS，53％的可能性NS和7％的可能NS（Zajicek标准）。抗肉芽肿/免疫抑制治疗方案各异，有31％死亡。结论：本系列扩大了我们对NS-ICH病理学的认识，NS-ICH可能起源于动脉或静脉。三分之一有孤立的NS。临床医生应在年轻的ICH患者中考虑NS，因为早期积极的抗颗粒疗法可能会改善结局。

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