Reversible cerebral vasoconstriction syndrome due to teprotumumab: two case reports.

IF 0.5 Q3 MEDICINE, GENERAL & INTERNAL

Oxford Medical Case Reports Pub Date : 2024-08-06 eCollection Date: 2024-08-01 DOI:10.1093/omcr/omae085

Mohamed Elfil, Pashayar P Lookian, Kanchan Kumari, Mohammad Aladawi, Mark Jedras, Steven M Phillips, Mithun G Sattur

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引用次数: 0

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.

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特普鲁单抗导致的可逆性脑血管收缩综合征：两例报告。

背景：可逆性脑血管收缩综合征（RCVS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 的确切病理生理学尚不完全清楚，但有多种病因被认为与引发 RCVS 有关。我们报告了两例可能与替普鲁单抗有关的 RCVS 病例。病例 1：一名 59 岁的女性患者患有巴塞杜氏眼病（GED），在开始使用替普鲁单抗后出现腿部无力和头痛，神经影像学检查发现其患有多灶性脑血管痉挛（CVS）。维拉帕米缓解了血管痉挛，患者总体病情有所好转。病例 2：一名 71 岁的女性 GED 患者在开始使用替普鲁单抗两个月后出现雷击样头痛，神经影像学检查显示为蛛网膜下腔出血（SAH）和 CVS。患者服用维拉帕米后病情好转，出院时未出现任何功能障碍：结论：开始使用替普鲁单抗与 RCVS 症状出现之间的时间相关性令人担忧替普鲁单抗可能会通过破坏脑血管调节而引发 RCVS。我们需要进一步研究这种关联。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Oxford Medical Case Reports MEDICINE, GENERAL & INTERNAL-

CiteScore

0.90

自引率

0.00%

发文量

125

审稿时长

19 weeks

期刊介绍： Oxford Medical Case Reports (OMCR) is an open access, peer-reviewed online journal publishing original and educationally valuable case reports that expand the field of medicine. The journal covers all medical specialities including cardiology, rheumatology, nephrology, oncology, neurology, and reproduction, comprising a comprehensive resource for physicians in all fields and at all stages of training. Oxford Medical Case Reports deposits all articles in PubMed Central (PMC). Physicians and researchers can find your work through PubMed , helping you reach the widest possible audience. The journal is also indexed in the Web of Science Core Collection . Oxford Medical Case Reports publishes case reports under the following categories: Allergy Audiovestibular medicine Cardiology and cardiovascular systems Critical care medicine Dermatology Emergency medicine Endocrinology and metabolism Gastroenterology and hepatology Geriatrics and gerontology Haematology Immunology Infectious diseases and tropical medicine Medical disorders in pregnancy Medical ophthalmology Nephrology Neurology Oncology Paediatrics Pain Palliative medicine Pharmacology and pharmacy Psychiatry Radiology, nuclear medicine, and medical imaging Respiratory disorders Rheumatology Sexual and reproductive health Sports Medicine Substance abuse.