Abstract:
BACKGROUND: Reversible Cerebral Vasoconstriction Syndrome (RCVS) involves cerebral vasculature constriction and dilation. While the exact pathophysiology of RCVS is still not fully understood, there are multiple etiological factors suggested to be implicated in triggering RCVS. We report two RCVS cases potentially linked to teprotumumab. Case 1: A 59-year-old female with Graves\' eye disease (GED) developed leg weakness and headache after initiating teprotumumab, and neuroimaging studies revealed multifocal cerebral vasospasm (CVS). Verapamil mitigated vasospasm and the patient overall improved. Case 2: A 71-year-old female with GED developed thunderclap headache two months after starting teprotumumab, with subarachnoid hemorrhage (SAH) and CVS revealed on neuroimaging studies. The patient improved on verapamil and was discharged without deficits.
CONCLUSIONS: The temporal correlation between teprotumumab initiation and RCVS\'s symptom onset raises concern for the potential involvement of teprotumumab in triggering RCVS via disrupting cerebrovascular modulation. Further research is needed to investigate this proposed association.
摘要:
背景:可逆性脑血管收缩综合征(RCVS)涉及脑血管收缩和扩张。虽然RCVS的确切病理生理学仍未完全了解,提示有多种病因与触发RCVS有关。我们报告了两例可能与teprotumumab相关的RCVS病例。病例1:一名患有Graves眼病(GED)的59岁女性在开始teprotumumab后出现腿部无力和头痛,神经影像学研究显示多灶性脑血管痉挛(CVS)。维拉帕米减轻血管痉挛,患者总体改善。病例2:一名71岁的GED女性在开始teprotumumab后两个月出现雷击性头痛,神经影像学研究显示蛛网膜下腔出血(SAH)和CVS。患者使用维拉帕米有所改善,出院时无缺陷。
结论:teprotumumab起始和RCVS症状发作之间的时间相关性引起了人们对teprotumumab通过破坏脑血管调节来触发RCVS的潜在参与的关注。需要进一步的研究来调查这种拟议的关联。