Finsterer Josef
Neurology, Neurology and Neurophysiology Center, Vienna, AUT.
Cureus. 2023 Dec 13;15(12):e50427. doi: 10.7759/cureus.50427. eCollection 2023 Dec.
Erdheim-Chester disease (ECD) is a histiocytosis that infiltrates all organs, but especially large arteries such as the aorta and its branches, which become sheathed and lead to multiple stenoses or even occlusions. Subclavian steal syndrome (SSS) has not been reported in ECD. A 68-year-old female was diagnosed with ECD due to the typical sheathing of the aorta and its major branches. Five years previously, asymptomatic SSS due to stenosis of the left subclavian artery was incidentally diagnosed. In the following years, occlusion of the subclavian and left vertebral artery, and stenosis of basilar artery and right middle cerebral artery occurred. The abnormal cerebral perfusion had consequences on the perfusion of the left upper extremity and was presumably responsible for falls. Basilar and middle cerebral artery stenosis is rare in ECD and vertebral artery occlusion and double subclavian steal have not been reported in ECD. This case is the first to show that the initial manifestation of ECD can be unilateral SSS and that subclavian artery occlusion can even lead to double SSS. Patients with SSS should undergo a thorough diagnostic evaluation to detect rare causes of SSS, such as ECD.
厄德里希-切斯特病(ECD)是一种组织细胞增多症,可浸润所有器官,尤其是主动脉及其分支等大动脉,这些动脉会被包裹,导致多处狭窄甚至闭塞。ECD患者中尚未有锁骨下动脉盗血综合征(SSS)的报道。一名68岁女性因主动脉及其主要分支出现典型的包裹现象而被诊断为ECD。五年前,该患者因左锁骨下动脉狭窄偶然被诊断出无症状性SSS。在接下来的几年里,锁骨下动脉和左椎动脉闭塞,基底动脉和右大脑中动脉狭窄。异常的脑灌注对左上肢灌注产生了影响,可能是导致跌倒的原因。基底动脉和大脑中动脉狭窄在ECD中较为罕见,ECD患者中尚未有椎动脉闭塞和双侧锁骨下动脉盗血的报道。该病例首次表明,ECD的初始表现可为单侧SSS,且锁骨下动脉闭塞甚至可导致双侧SSS。患有SSS的患者应进行全面的诊断评估,以检测SSS的罕见病因,如ECD。