Department of Neurology, King Fahd University Hospital, Imam Abdulrahman Bin Faisal University, Al Khobar, Saudi Arabia.
College of Medicine, King Fahd University Hospital, Imam Abdulrahman Bin Faisal University, Al Khobar, Saudi Arabia.
Am J Case Rep. 2021 Nov 16;22:e934528. doi: 10.12659/AJCR.934528.
BACKGROUND Reversible cerebral vasoconstriction syndrome (RCVS) is a rare neurological disorder with a complex physiopathology that is not fully understood. Suggested underlying mechanisms include failure of autoregulation, endothelial dysfunction, and oxidative stress. It is characterized by reversible multifocal constriction of the cerebral arteries, and can be triggered by many conditions, including, vasoactive medications (eg, triptans), cerebrovascular events, primary headache disorders, and metabolic causes (eg, hypercalcemia). RCVS can also be associated with pregnancy-related conditions, such as thrombotic thrombocytopenic purpura, eclampsia, and pre-eclampsia. Thunderclap headache is the most common clinical manifestation; however, other symptoms can result from complications of the disease, such as stroke, brain edema, and seizures. Several case reports have been published of an association between RCVS and eclampsia, but to the best of our knowledge, only 3 cases were successfully treated with intravenous milrinone and this is the only patient reported in Saudi Arabia. CASE REPORT We report a case of 25-year-old primigravida woman who presented with acute-onset headache, nausea, elevated blood pressure, and generalized tonic clonic seizure. She was diagnosed as having RCVS secondary to eclampsia based on clinical and radiological features. She was initially started on nimodipine, which is usually the first-line management of RCVS, as well as magnesium sulfate and levetiracetam; however, she only achieved full recovery after starting intravenous milrinone. CONCLUSIONS Milrinone is one of the emerging drugs for treatment of RCVS, and this case report delineates the potential of using the drug, especially in cases refractory to standard therapy.
可逆性脑血管收缩综合征(RCVS)是一种罕见的神经疾病,其复杂的病理生理学尚未完全阐明。潜在的机制包括自动调节失败、内皮功能障碍和氧化应激。其特征是脑动脉的多灶性可逆收缩,并可由多种情况引发,包括血管活性药物(如曲坦类)、脑血管事件、原发性头痛疾病和代谢原因(如高钙血症)。RCVS 也可能与妊娠相关疾病相关,如血栓性血小板减少性紫癜、子痫和子痫前期。霹雳性头痛是最常见的临床表现;然而,其他症状可能是由于疾病的并发症引起的,如中风、脑水肿和癫痫发作。已经发表了几篇关于 RCVS 与子痫的病例报告,但据我们所知,只有 3 例成功地用米力农静脉治疗,这也是沙特阿拉伯唯一报告的病例。
我们报告了一例 25 岁初产妇,表现为突发头痛、恶心、血压升高和全身强直阵挛性癫痫发作。根据临床和影像学特征,她被诊断为子痫继发 RCVS。她最初开始使用尼莫地平,这通常是 RCVS 的一线治疗方法,以及硫酸镁和左乙拉西坦;然而,在开始使用米力农静脉滴注后,她才完全康复。
米力农是治疗 RCVS 的新兴药物之一,本病例报告阐述了该药物的潜在用途,尤其是在对标准治疗无反应的情况下。