Choi Maxwell C Y, Law Tiffany H P, Chen Sirong, Cheung William S K, Yim Carmen, Ng Oliver K S, Au Lisa W C, Mok Vincent C T, Woo Peter Y M
Department of Neurosurgery, Kwong Wah Hospital, Kowloon, Hong Kong SAR, China.
Research Department, Hong Kong Sanatorium and Hospital, Hong Kong, Hong Kong SAR, China.
Front Neurol. 2024 Mar 19;15:1360705. doi: 10.3389/fneur.2024.1360705. eCollection 2024.
Cases of iatrogenic cerebral amyloid angiopathy (CAA) have been increasingly reported recently, particularly those associated with neurosurgery. Preclinical studies have shown taxifolin to be promising for treating CAA. We describe a young 42-year-old man with a history of childhood traumatic brain injury that required a craniotomy for hematoma evacuation. He later presented with recurrent lobar intracerebral hemorrhage (ICH) decades later, which was histologically confirmed to be CAA. Serial C-Pittsburgh compound B positron emission tomography (C-PiB-PET) imaging showed a 24% decrease in global standardized uptake value ratio (SUVR) at 10 months after taxifolin use. During this period, the patient experienced clinical improvement with improved consciousness and reduced recurrent ICH frequency, which may be partly attributable to the potential amyloid-β (Aβ) clearing the effect of taxifolin. However, this effect seemed to have diminished at 15 months, CAA should be considered in young patients presenting with recurrent lobar ICH with a history of childhood neurosurgery, and serial C-PiB-PET scans warrant further validation as a strategy for monitoring treatment response in CAA for candidate Aβ-clearing therapeutic agents such as taxifolin.
近年来,医源性脑淀粉样血管病(CAA)的病例报告日益增多,尤其是与神经外科手术相关的病例。临床前研究表明,花旗松素在治疗CAA方面具有前景。我们描述了一名42岁的年轻男性,他有儿童期创伤性脑损伤病史,曾因血肿清除接受开颅手术。数十年后,他出现复发性脑叶脑出血(ICH),经组织学证实为CAA。连续的C-匹兹堡化合物B正电子发射断层扫描(C-PiB-PET)成像显示,使用花旗松素10个月后,整体标准化摄取值比率(SUVR)下降了24%。在此期间,患者的意识改善,复发性ICH频率降低,临床症状有所改善,这可能部分归因于花旗松素潜在的淀粉样β蛋白(Aβ)清除作用。然而,这种效果在15个月时似乎有所减弱,对于有儿童期神经外科手术史且出现复发性脑叶ICH的年轻患者,应考虑CAA的可能性,连续的C-PiB-PET扫描作为监测CAA中候选Aβ清除治疗药物(如花旗松素)治疗反应的策略,有待进一步验证。