Neurosurgery Department, Sheffield Teaching Hospitals NHS Foundation Trust, Sheffield, UK
Neurosurgery Department, Sheffield Teaching Hospitals NHS Foundation Trust, Sheffield, UK.
BMJ Case Rep. 2024 May 27;17(5):e259452. doi: 10.1136/bcr-2023-259452.
Fragile X-associated tremor/ataxia syndrome (FXTAS) is a progressive hereditary neurodegenerative disorder which causes intention tremor and cerebellar ataxia. It typically affects the ageing population. Deep brain stimulation (DBS) is widely accepted in the treatment of common movement disorders and has been trialled in treating rare and complex neurodegenerative disorders. We report a case of a man in his 40s with a long history of tremor affecting his hands. MRI brain revealed high T2 signal in the middle cerebellar peduncles. Genetic testing revealed FMR1 premutation confirming the diagnosis of FXTAS. Subsequently, he was treated with multitarget DBS of the ventralis intermediate nucleus and ventralis oralis posterior nuclei bilaterally, with excellent neurological function at 9 years follow-up. This case suggests multitarget DBS for FXTAS with neurophysiology-guided DBS programming can provide excellent long-term tremor suppression in selected patients.
脆性 X 相关震颤/共济失调综合征(FXTAS)是一种进行性遗传性神经退行性疾病,可导致意向性震颤和小脑共济失调。它通常影响老年人群。深部脑刺激(DBS)广泛应用于常见运动障碍的治疗,并已在治疗罕见和复杂神经退行性疾病方面进行了试验。我们报告了一例 40 多岁男性,手部震颤病史较长。脑 MRI 显示中脑小脑脚高 T2 信号。基因检测显示 FMR1 前突变,确诊为 FXTAS。随后,他接受了双侧腹侧中间核和腹侧前核多靶点 DBS 治疗,9 年随访时神经功能良好。该病例提示,神经生理学引导的 DBS 程控多靶点 DBS 可为选定患者提供优异的长期震颤抑制。