Lu Xiaocheng, Fang Xinggen, Huang Yabo, Zhou Peng, Wang Zhong, Brinjikji Waleed, Chen Gang
Department of Neurosurgery, The First Affiliated Hospital of Soochow University, Suzhou, China.
Department of Neurosurgery, Wannan Medical College, Yijishan Hospital, Wuhu, China.
Front Neurol. 2021 Dec 16;12:755312. doi: 10.3389/fneur.2021.755312. eCollection 2021.
Pure arterial malformations (PAMs) are extremely rare abnormalities defined as dilated, overlapping, and tortuous arteries with a coil-like appearance in the absence of venous components. Over the last half century, only seven published reports have described cases of patients with PAMs who received treatment. Here, we report two cases of women with PAMs who received surgical treatment, and we present a systematic review of the literature. We searched the PubMed, Embase, Web of Science, and Medline databases (up until October 1, 2021) for relevant publications. We performed independent-sample -tests and Fisher's exact tests to compare continuous and categorical characteristics among the available cases. Our first patient was a 43-year-old woman with PAM of the left internal carotid artery (ICA), who received an ICA-radial artery (RA)-M2 bypass. Post-operative digital subtraction angiography (DSA) revealed the disappearance of the left ICA PAM without ischemic events during follow-up. The second patient was a 53-year-old woman with PAMs of the right ICA and posterior cerebral artery. The P1 lesion was treated by proximal occlusion combined with a superficial temporal artery-P2 bypass. During the 12-month follow-up period, the size of the PAMs decreased significantly as indicated by the post-operative DSA showing the absence of hemorrhages. Our systematic review, which includes 56 PAMs, shows that the reported PAMs were more common in the anterior circulation (33/56, 58.9%) than in the posterior circulation (11/56, 19.7%). Bilateral PAMs were more likely to affect bilateral anterior cerebral arteries (ACA) (ACA vs. ACA: 63.6 vs. 26.2%, = 0.02). In addition, PAMs involving the anterior circulation were likely to affect multiple arteries (anterior vs. posterior: 30.3 vs. 0%, = 0.038). We found very few reports on treated PAMs; further studies with large sample sizes and long follow-up periods are required to explore the appropriate treatment strategy for PAMs.
纯动脉畸形（PAMs）是极为罕见的异常情况，定义为在没有静脉成分的情况下，动脉呈扩张、重叠且迂曲的螺旋状外观。在过去的半个世纪里，仅有七篇发表的报告描述了接受治疗的PAMs患者病例。在此，我们报告两例接受手术治疗的患有PAMs的女性病例，并对相关文献进行系统综述。我们在PubMed、Embase、Web of Science和Medline数据库（截至2021年10月1日）中搜索相关出版物。我们进行独立样本t检验和Fisher精确检验，以比较现有病例中的连续和分类特征。我们的首例患者是一名43岁女性，患有左颈内动脉（ICA）的PAM，接受了ICA-桡动脉（RA）-M2搭桥手术。术后数字减影血管造影（DSA）显示左ICA PAM消失，随访期间未发生缺血事件。第二例患者是一名53岁女性，患有右ICA和大脑后动脉的PAMs。P1病变通过近端闭塞联合颞浅动脉-P2搭桥进行治疗。在12个月的随访期内，PAMs的大小显著减小，术后DSA显示无出血。我们的系统综述纳入了56例PAMs，结果显示报告的PAMs在前循环（33/56，58.9%）比后循环（11/56，19.7%）更常见。双侧PAMs更有可能累及双侧大脑前动脉（ACA）（ACA与ACA：63.6%对2*6.2%，P = 0.02）。此外，累及前循环的PAMs有可能累及多条动脉（前循环与后循环：30.3%对0%，P = 0.038）。我们发现关于接受治疗的PAMs的报告非常少；需要进行更大样本量和更长随访期的进一步研究，以探索PAMs的合适治疗策略。
