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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.

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。

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