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酷似囊性血管瘤病的巨大颅面骨内血管畸形:2例报告并文献复习

Massive craniofacial intraosseous vascular malformation resembling cystic angiomatosis: report of 2 cases and review of the literature.

作者信息

Hossein Mortazavi Seyed, Khodayari Abbas, Khojasteh Arash, Abbas Fatemeh Mashhadi, Mehrdad Leili, Kiani Mohammad Taghi, Farman Allan G

机构信息

Department of Oral and Maxillofacial Surgery, Shahid Beheshti University of Medical Sciences, Tehran, Iran.

出版信息

J Oral Maxillofac Surg. 2011 Jan;69(1):204-14. doi: 10.1016/j.joms.2010.07.072. Epub 2010 Nov 2.

DOI:10.1016/j.joms.2010.07.072
PMID:21050648
Abstract

Cystic angiomatosis of bone is a rare condition of multifocal angiomas of the skeleton. The condition is believed to be congenital, grows slowly and starts in first decades of life. Two cases of progressive bimaxillary enlargement, presented here with a history of slowly enlargement of facial bones when they were 9 and 6 year old, respectively. Radiographic evaluation of the craniofacial bones revealed aggressive hypertrophy with severe displacement of the teeth. The histopathological evaluation of the gross specimen showed vital bone containing capillary and cavernous spaces with endothelial lining. Aggressive cystic angiomatosis of the facial bones was described here as the most probable diagnosis.

摘要

骨囊性血管瘤病是一种罕见的骨骼多灶性血管瘤疾病。该病被认为是先天性的,生长缓慢,发病于生命的最初几十年。本文报告两例进行性双上颌骨增大病例,分别为9岁和6岁,有面部骨骼缓慢增大的病史。颅面骨的影像学评估显示有侵袭性肥大,牙齿严重移位。大体标本的组织病理学评估显示,活骨内含有有内皮衬里的毛细血管和海绵状间隙。本文将面部骨骼的侵袭性囊性血管瘤病描述为最可能的诊断。

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Vascular Floor of Mouth Mass with Unanticipated Intracranial, Orbital, and Vertebral Associated Involvements.伴有意外颅内、眶内及椎骨相关累及的口腔底部血管性肿物
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Loss-of-Function Mutations in ELMO2 Cause Intraosseous Vascular Malformation by Impeding RAC1 Signaling.
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