• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

一名11个月大患者的后颅窝巨大海绵状畸形伴淋巴管瘤样表型,与眼眶静脉淋巴管异常相关:病例报告及文献综述

Giant cavernous malformation of the posterior fossa with lymphangiomatous phenotype, associated with orbital venolymphatic anomaly in an 11-month-old patient: case report and literature review.

作者信息

Flores-Sanchez Jose Daniel, Pregúntegui Ivethe, Ugas Carlos, Cruzado Carla, Ramirez Alberto, Poterico Julio A

机构信息

Department of Pediatric Neurosurgery, Instituto Nacional de Salud del Niño San Borja, Lima, Peru.

Department of Radiology, Instituto Nacional de Salud del Niño San Borja, Lima, Peru.

出版信息

Childs Nerv Syst. 2023 Jan;39(1):289-293. doi: 10.1007/s00381-022-05623-3. Epub 2022 Jul 29.

DOI:10.1007/s00381-022-05623-3
PMID:35904587
Abstract

The synchronous presentation of venolymphatic anomalies of the orbit and noncontiguous intracranial cavernous malformations is uncommon. Herein, we present a case of an 11-month-old female patient diagnosed with orbital venolymphatic anomaly associated with a large cavernous malformation in the posterior fossa, who underwent complete surgical resection of the latter. The immunohistochemical analysis was positive for podoplanin, a marker expressed by lymphatic endothelial cells, but not vascular endothelium. This exceptional finding suggests lymphatic involvement in the etiology of the lesion. In our review of the literature, we did not find similar cases in patients under 1 year of age.

摘要

眼眶静脉淋巴管畸形与非相邻颅内海绵状血管畸形同时出现的情况并不常见。在此,我们报告一例11个月大的女性患者,诊断为眼眶静脉淋巴管畸形,伴有后颅窝大型海绵状血管畸形,该患者接受了后者的完全手术切除。免疫组织化学分析显示,淋巴管内皮细胞表达的标志物血小板内皮细胞黏附分子-1呈阳性,而血管内皮细胞不表达。这一特殊发现提示淋巴管参与了病变的病因。在我们的文献回顾中,未发现1岁以下患者有类似病例。

相似文献

1
Giant cavernous malformation of the posterior fossa with lymphangiomatous phenotype, associated with orbital venolymphatic anomaly in an 11-month-old patient: case report and literature review.一名11个月大患者的后颅窝巨大海绵状畸形伴淋巴管瘤样表型,与眼眶静脉淋巴管异常相关:病例报告及文献综述
Childs Nerv Syst. 2023 Jan;39(1):289-293. doi: 10.1007/s00381-022-05623-3. Epub 2022 Jul 29.
2
Combined venous lymphatic malformations of the orbit (so-called lymphangiomas). Association with noncontiguous intracranial vascular anomalies.眼眶静脉淋巴管混合性畸形(所谓的淋巴管瘤)。与非连续性颅内血管异常相关。
Ophthalmology. 1998 Jan;105(1):176-84. doi: 10.1016/s0161-6420(98)92058-9.
3
Novel treatment of recurrent orbital venolymphatic malformation with sirolimus and rivaroxaban.西罗莫司和利伐沙班治疗复发性眼眶静脉淋巴管畸形的新方法。
Orbit. 2022 Dec;41(6):771-778. doi: 10.1080/01676830.2021.1929339. Epub 2021 May 25.
4
Multi-compartment skull base orbital cavernous venous malformation: A rare presentation of a common orbital mass.多房性颅底眼眶海绵状静脉畸形:一种常见眼眶肿物的罕见表现。
Am J Ophthalmol Case Rep. 2021 Jan 21;21:101020. doi: 10.1016/j.ajoc.2021.101020. eCollection 2021 Mar.
5
Giant, symptomatic mixed vascular malformation containing a cavernoma, developmental venous anomaly, and capillary telangiectasia in a 19-month-old infant.一名19个月大婴儿患有巨大的、有症状的混合性血管畸形,包含海绵状血管瘤、静脉发育异常和毛细血管扩张症。
Childs Nerv Syst. 2022 May;38(5):1005-1009. doi: 10.1007/s00381-021-05358-7. Epub 2021 Sep 30.
6
Distensible venous malformations of the orbit: clinical and hemodynamic features and a new technique of management.眼眶可扩张性静脉畸形:临床与血流动力学特征及一种新的治疗技术
Ophthalmology. 1999 Jun;106(6):1197-209. doi: 10.1016/S0161-6420(99)90245-2.
7
Orbital lymphangioma with positive immunohistochemistry of lymphatic endothelial markers (vascular endothelial growth factor receptor 3 and podoplanin).眼眶淋巴管瘤,淋巴管内皮标志物(血管内皮生长因子受体3和足板蛋白)免疫组化呈阳性。
Graefes Arch Clin Exp Ophthalmol. 2001 Aug;239(8):628-32. doi: 10.1007/s004170100325.
8
Venolymphatic vascular malformation of the parotid gland extending into the parapharyngeal space: A rare presentation.腮腺静脉淋巴血管畸形延伸至咽旁间隙:一种罕见的表现。
J Oral Maxillofac Pathol. 2016 May-Aug;20(2):308-11. doi: 10.4103/0973-029X.185917.
9
Intracranial vascular anomalies in patients with periorbital lymphatic and lymphaticovenous malformations.眶周淋巴管瘤和淋巴管静脉瘤患者的颅内血管异常
AJNR Am J Neuroradiol. 2007 Feb;28(2):335-41.
10
Dural sinus malformation with giant pouch (DSMGP): symptoms and treatment.伴有巨大囊袋的硬脑膜窦畸形(DSMGP):症状与治疗
Childs Nerv Syst. 2020 Feb;36(2):343-348. doi: 10.1007/s00381-019-04338-2. Epub 2019 Aug 8.

本文引用的文献

1
Meningeal lymphatic vessels regulate brain tumor drainage and immunity.脑膜淋巴管调节脑肿瘤引流和免疫。
Cell Res. 2020 Mar;30(3):229-243. doi: 10.1038/s41422-020-0287-8. Epub 2020 Feb 24.
2
Current understanding of lymphatic vessels in the central nervous system.目前对中枢神经系统淋巴管的认识。
Neurosurg Rev. 2020 Aug;43(4):1055-1064. doi: 10.1007/s10143-019-01133-0. Epub 2019 Jun 18.
3
The Meningeal Lymphatic System: A New Player in Neurophysiology.脑膜淋巴系统:神经生理学的新角色。
Neuron. 2018 Oct 24;100(2):375-388. doi: 10.1016/j.neuron.2018.09.022.
4
Cerebellar cavernous malformation in pediatric patients: defining clinical, neuroimaging, and therapeutic characteristics.小儿患者的小脑海绵状血管畸形:界定临床、神经影像学及治疗特征
J Neurosurg Pediatr. 2015 Sep;16(3):256-66. doi: 10.3171/2015.1.PEDS14366. Epub 2015 May 22.
5
Occasional finding of a dural arteriovenous fistula in a newborn with orbital lymphangioma. A case report.
Neuroradiol J. 2010 Apr;23(2):191-4. doi: 10.1177/197140091002300207. Epub 2010 Apr 20.
6
Vascular anomalies of the head and neck: a review of genetics.头颈部血管异常:遗传学综述
Semin Ophthalmol. 2013 Sep-Nov;28(5-6):257-66. doi: 10.3109/08820538.2013.825279. Epub 2013 Sep 6.
7
Intracranial cavernous malformation in children: a single-centered experience with 30 consecutive cases.儿童颅内海绵状畸形:30例连续病例的单中心经验
Arq Neuropsiquiatr. 2013 Apr;71(4):220-8. doi: 10.1590/0004-282x20130006.
8
Value of podoplanin as an immunohistochemical marker in tumor diagnosis: a review and update.足突蛋白作为肿瘤诊断中免疫组化标志物的价值:综述与更新
Appl Immunohistochem Mol Morphol. 2014 May-Jun;22(5):331-47. doi: 10.1097/PAI.0b013e31828a83c5.
9
Cavernous malformations: a paradigm for progress.
Clin Neurosurg. 2011;58:27-41. doi: 10.1227/neu.0b013e318226a069.
10
Pediatric cavernous malformation in the central nervous system: report of 66 cases.小儿中枢神经系统海绵状畸形:66例报告。
Pediatr Neurosurg. 2009;45(2):105-13. doi: 10.1159/000209284. Epub 2009 Mar 21.