猎人弓综合征病例报告及系统综述

A case of bow hunter's syndrome and a systematic literature review

目的:报告1例寰椎左侧骨赘导致旋转性椎动脉闭塞(猎人弓综合征,bow hunter’s syndrome,BHS)的临床病例,进一步分析BHS的流行病学特征、诊疗方式及其临床结局。方法:对上述提到的1例BHS患者的临床资料、诊断方法、治疗方式和临床结局进行描述。检索1978至2021年的文献,将所涉及的BHS患者作为研究对象,收集每例入选患者的发病年龄、性别、病因、发病部位、诊断方法、治疗方式及临床结局等资料。采用系统分析的方法对数据进行分项研究。结果:报告1例因寰椎左侧骨赘导致左侧椎动脉旋转性闭塞的患者,三维CT重建显示寰椎左侧骨赘压迫椎动脉;动态数字减影血管造影显示左侧椎动脉V2段远端轻度狭窄,向左侧转头时该部分椎动脉受到卡压导致血流中断,头部恢复中立位后血流通畅。因保守治疗后症状无法缓解,采用后路寰椎骨赘切除术+椎动脉减压术,术后患者症状基本消失,短期随访结果良好。筛选出的87篇文献及报告的1例患者共126例BHS患者纳入研究。发病中位年龄55岁(43.5岁,65.0岁),高发年龄51~60岁;男女比例为1.9∶1。126例患者中有65例患者椎动脉闭塞或者狭窄的部位位于寰枢椎节段;66例患者累及左侧椎动脉,45例患者累及右侧椎动脉,15例患者累及双侧椎动脉。诊断方法:126例患者中有114例患者均采用全脑动态数字减影血管造影(dynamic digital subtraction angiography,dDSA)来确诊BHS。随访时间0.25~114月,平均16.6月。治疗方式及临床疗效:36例患者采取保守治疗,其中12例患者治疗后存在残余症状;32例患者采取融合手术,所有患者术后症状均得到缓解;54例患者采取单纯减压手术,其中4例患者术后存在残余症状;4例患者接受介入治疗,术后症状均得到缓解。结论:BHS患者临床少见,常累及寰枢椎节段,左侧椎动脉较易受累,高发年龄为51~60岁,DSA是诊断BHS的金标准,对于由异常纤维骨结构、椎间盘突出、节段不稳定等原因导致的BHS,椎动脉或寰枢椎融合的减压手术是最常用的治疗方式。

Objective A clinical case of rotational vertebral artery occlusion(bow hunter's syndrome,BHS)caused by left C1 osteophyte was reported,and the epidemiological characteristics,diagnosis and treatment methods,and clinical outcomes of BHS were further analyzed.Methods The clinical data,diagnostic methods,treatment options,and clinical outcomes of the above-mentioned BHS patient are described.The literature from 1978 to 2021 was retrieved,the BHS patients involved were taken as the research objects,and the data of onset age,gender,etiology,site of onset,diagnosis method,treatment method,and clinical outcomes of each selected patient were collected.The data were subdivided through systematic analysis.Results A patient with rotational compression of the left vertebral artery associated with the left osteophyte of the atlas was presented.3D-CT showed that the vertebral artery was compressed by the left osteophyte of the atlas.Dynamic digital subtraction angiography(dDSA)showed mild stenosis of the distal V2 segment of the left vertebral artery.When his head turned to the left,the distal V2 segment of the left vertebral artery was compressed and the blood flow was interrupted.After his head was in a neutral position,the blood flow was restored.Because the symptoms could not be relieved after conservative treatment,posterior C1 osteophyte resection was used to decompress the vertebral artery,and the symptoms disappeared after the operation,and the short-term follow-up results were good.All of the 87 articles and 126 patients have been studied.The median age was 55.0 years(IQR:43.5,65.0 years)and the peak age of onset was 51 to 60 years old.The gender difference has been uncovered and the sex ratio was 1.9∶1(male∶female).Among the 126 patients,65 patients had vertebral artery occlusion or stenosis located in the C1-C2 segment;66 patients involved the left vertebral artery,45 patients involved the right vertebral artery,and 15 patients involved bilateral vertebral arteries.DSA was used to confirm BHS in 114 of 126 patients.The follow-up time was 0.25-114 months,with an average of 16.6 months.Thirty-six patients were treated conservatively,and 12 patients had residual symptoms;33 patients underwent fusion surgery,and all patients'symptoms were relieved after surgery;54 patients underwent simple decompression surgery,and 4 patients had residual symptoms after surgery;4 patients received endovascular surgery,and their symptoms were relieved after surgery.Conclusion Patients with BHS are rare clinically,often involving C1-C2 and the left vertebral artery is more likely to be involved.The peak age of onset was 51 to 60 years old.DSA is the gold standard for the diagnosis of BHS.For BHS caused by abnormal bone structure,intervertebral disc herniation,joint instability,etc.,decompressive surgery of the vertebral artery or C1-C2 segment fusion is the most common treatment modality.