血友病性假肿瘤摘除术后上颌骨骨内静脉畸形：一例报告。
Intraosseous venous malformation of the maxilla after enucleation of a hemophilic pseudotumor: A case report.
作者信息
Cai Xu, Yu Jian-Jun, Tian Hao, Shan Zhen-Feng, Liu Xiao-Yu, Jia Jun
机构信息
Department of Head & Neck Surgery, Hunan Cancer Hospital & The Affiliated Cancer Hospital of Xiangya School of Medicine, Central South University, Changsha 410000, Hunan Province, China.
Physical Examination Center, The First Hospital of Changsha, Changsha 410000, Hunan Province, China.
出版信息
World J Clin Cases. 2020 Oct 6;8(19):4644-4651. doi: 10.12998/wjcc.v8.i19.4644.
BACKGROUND
Hemophilic pseudotumor (HP) is a rare complication in patients with hemophilia. The lesion most frequently occurs in the long bones, pelvis, small bones of the hands and feet, or rarely in the maxillofacial region. Postoperative changes in HP are seldom arrested, whereas angiogenesis characterized by disturbed wound healing in HP may cause vascular malformations.
CASE SUMMARY
We report the case of an 11-year-old boy who was affected by maxillary intraosseous venous malformation. Enucleation of an HP without factor replacement was performed initially on the right side of the maxilla 3 years ago. The patient was referred to us because of painless swelling in the same location. Factor replacement and subtotal maxillectomy were performed. Pathological examinations revealed intraosseous venous malformation.
CONCLUSION
This study is the first to document the development of intraosseous venous malformation after enucleation of an HP in the maxillofacial region. Angiogenesis characterized by disturbed wound healing in patients with hemophilia may be pivotal in the pathogenesis of this condition.
背景
血友病性假肿瘤（HP）是血友病患者的一种罕见并发症。该病变最常发生于长骨、骨盆、手足小骨，极少发生于颌面区域。HP术后改变很少停止，而以HP伤口愈合障碍为特征的血管生成可能导致血管畸形。
病例摘要
我们报告一例11岁患上颌骨内静脉畸形的男孩。3年前首次在右侧上颌骨行HP摘除术，未进行因子替代治疗。患者因同一部位无痛性肿胀前来就诊。进行了因子替代治疗及上颌骨次全切除术。病理检查显示骨内静脉畸形。
结论
本研究首次记录了颌面区域HP摘除术后骨内静脉畸形的发生情况。以血友病患者伤口愈合障碍为特征的血管生成可能在该病的发病机制中起关键作用。
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