Zhang Jinwei,Wu Shuihua,Su Jun,et al.Management of vein of Galen aneurysmal malformation with interventional embolization in infants and children: a report of 2 cases with a literature review[J].Journal of Clinical Pediatric Surgery,2025,(02):131-137.[doi:10.3760/cma.j.cn101785-202410066-006]
介入栓塞治疗婴幼儿Galen静脉动脉瘤样畸形2例并文献综述
- Title:
- Management of vein of Galen aneurysmal malformation with interventional embolization in infants and children: a report of 2 cases with a literature review
- Keywords:
- Intracranial Arteriovenous Malformations; Vein Aneurysmal of Galen Malformation; Endovascular Therapy; Embolization; Therapeutic; Child
- 摘要:
- 目的 探讨婴幼儿Galen静脉动脉瘤样畸形的临床特征、诊断及介入栓塞治疗的效果。方法 回顾性分析2020年1月至2022年12月湖南省儿童医院神经外科收治的2例Galen静脉动脉瘤样畸形患儿临床资料,并检索2014年1月至2024年10月PubMed、万方医学网和中国知网中介入栓塞治疗婴幼儿Galen静脉动脉瘤样畸形相关文献,总结分析患儿临床特征、分型、诊断、介入栓塞治疗方式及治疗结果。结果 本院收治的2例中,病例1术后12个月头围52 cm,语言和大运动等发育较术前明显改善,能自行走路,复查头颅MRI提示Galen静脉动脉瘤样畸形较术前明显缩小。病例2术后6个月头围53.5 cm,语言发育较术前改善,复查头颅MRI提示Galen静脉动脉瘤样畸形较术前无变化,脑室扩张较前好转,家属拒绝再次行脑血管造影及介入栓塞治疗,目前已失访。共检索到符合纳排标准文献10篇,共报道介入栓塞治疗婴幼儿Galen静脉动脉瘤样畸形15例,加上本院2例,共17例。其中脉络膜型12例,壁内型5例;孕期B超检出3例;经静脉介入栓塞1例,经动静脉联合栓塞1例,经动脉介入栓塞15例。1例死亡,3例动脉瘤样畸形残留,13例治愈(76.47%)。在术前合并脑积水的11例患儿中,术后完全缓解7例、明显缓解2例、无缓解1例、死亡1例。结论 Galen静脉动脉瘤样畸形是一种罕见的颅内血管畸形,主要临床表现为头围增大、发育落后、心力衰竭,产前彩超、胎儿磁共振检查是产前诊断的主要手段,脑血管造影是确诊的金标准,介入栓塞治疗效果良好。
- Abstract:
- Objective To explore the clinical features,diagnoses and outcomes of interventional embolization for vein of Galen aneurysmal malformation (VGAM) in infants and toddlers. Methods Retrospective review was conducted for the relevant clinical data of two children of VGAM from January 2020 to December 2022.And the literature related to the treatment of VGAM with interventional embolization was searched in PubMed,Wanfang Medical Network and China Knowledge Network from January 2014 to October 2024.Clinical characteristics,stages,diagnoses,treatment protocols and treatment outcomes were summarized. Results Case 1 had a head circumference of 52 cm during a 12-month follow-up period.Development of speech and gross motor improved significantly as compared with preoperative period.Independent ambulation was feasible.Cranial magnetic resonance imaging (MRI) indicated that VGAM significantly diminished as compared with preoperative period.In case 2,head circumference was 53.5 cm during a 12-month follow-up period.Development of speech and language improved as compared with preoperative period.Cranial MRI revealed that VGAM had no change as compared with preoperative period and dilatation of cerebral ventricles improved as compared with preoperative period.The family declined to undergo cerebral angiography and interventional embolization again.The patient was lost to follow-ups.Ten articles retrieved from the literature fulfilled the inclusion and exclusion criteria.A total of 15 cases of VGAM in infants and children treated by interventional embolization were reported along with 2 cases at our hospital.The clinical types were choroidal (n=12) and intramural (n=5).Three cases were detected by ultrasound during pregnancy.The procedures included interventional embolization via venous (n=1),combined embolization via arterial-vein embolization (n=1) and trans-arterial interventional embolization (n=15).There were death (n=1) and residual aneurysm-like malformation (n=3).Curative rate was 76.47%.There were preoperative combined hydrocephalus (n=11),postoperative complete relief (n=7),obvious relief (n=2) and non-relief (n=1). Conclusions VGAM is a very rare intracranial vascular malformation with major clinical manifestations of expanded head circumference,growth retardation and heart failure.Intrapartum ultrasound and fetal magnetic resonance may aid in intrapartum diagnosis.And cerebral angiography is a gold standard for confirming a diagnosis.Interventional embolization is highly efficacious.
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备注/Memo
收稿日期:2024-10-29。
基金项目:湖南省自然科学基金资助项目(2025JJ50682)
通讯作者:吴水华,Email:292454021@qq.com