Elfil Mohamed, Lookian Pashayar P, Kumari Kanchan, Aladawi Mohammad, Jedras Mark, Phillips Steven M, Sattur Mithun G
Department of Neurological Sciences, University of Nebraska Medical Center, 988440 Nebraska Medical Center, Omaha, NE 68198-8440, United States.
Department of Neurosurgery, University of Nebraska Medical Center, 4242 Farnam St., Omaha, NE 68131, United States.
Oxf Med Case Reports. 2024 Aug 6;2024(8):omae085. doi: 10.1093/omcr/omae085. eCollection 2024 Aug.
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.
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.
可逆性脑血管收缩综合征（RCVS）涉及脑血管的收缩和扩张。虽然RCVS的确切病理生理学仍未完全了解，但有多种病因被认为与触发RCVS有关。我们报告了两例可能与替普罗单抗相关的RCVS病例。病例1：一名59岁患有格雷夫斯眼病（GED）的女性在开始使用替普罗单抗后出现腿部无力和头痛，神经影像学研究显示有多发性脑血管痉挛（CVS）。维拉帕米减轻了血管痉挛，患者总体情况有所改善。病例2：一名71岁患有GED的女性在开始使用替普罗单抗两个月后出现霹雳样头痛，神经影像学研究显示有蛛网膜下腔出血（SAH）和CVS。患者使用维拉帕米后病情改善，出院时无功能缺损。
替普罗单抗开始使用与RCVS症状发作之间的时间相关性引发了对替普罗单抗通过破坏脑血管调节触发RCVS的潜在可能性的担忧。需要进一步研究来调查这种拟议的关联。
