Case Report
Copyright ©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Oct 6, 2020; 8(19): 4644-4651
Published online Oct 6, 2020. doi: 10.12998/wjcc.v8.i19.4644
Intraosseous venous malformation of the maxilla after enucleation of a hemophilic pseudotumor: A case report
Xu Cai, Jian-Jun Yu, Hao Tian, Zhen-Feng Shan, Xiao-Yu Liu, Jun Jia
Xu Cai, Jian-Jun Yu, Hao Tian, Zhen-Feng Shan, 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
Xiao-Yu Liu, Physical Examination Center, The First Hospital of Changsha, Changsha 410000, Hunan Province, China
Jun Jia, The State Key Laboratory Breeding Base of Basic Science of Stomatology (Hubei-MOST) and Key Laboratory of Oral Biomedicine Ministry of Education, School and Hospital of Stomatology, Wuhan University, Wuhan 430000, Hubei Province, China
Author contributions: Cai X and Jia J were the patient’s maxillofacial surgeons and contributed to manuscript drafting; Tian H reviewed the literature and contributed to manuscript writing; Shan ZF contributed to collecting the clinical data; Liu XY contributed to immunohistochemical research; Yu JJ contributed to pathological research; all authors issued final approval for the version to be submitted.
Supported by the Key Clinical Specialty Construction Project (Head and Neck Surgery) of Provincial Health Commission of Hunan Province.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and accompanying images.
Conflict-of-interest statement: All authors declare that they have no conflict of interest to disclose.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Corresponding author: Xu Cai, DDS, MD, Attending Doctor, Department of Head and Neck Surgery, Hunan Cancer Hospital & The Affiliated Cancer Hospital of Xiangya School of Medicine, Central South University, No. 237 Tongzipo Road, Changsha 410000, Hunan Province, China. caixu@hnca.org.cn
Received: March 31, 2020
Peer-review started: March 31, 2020
First decision: April 28, 2020
Revised: May 11, 2020
Accepted: August 25, 2020
Article in press: August 25, 2020
Published online: October 6, 2020
Abstract
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.

Keywords: Hemophilia, Hemophilic pseudotumor, Maxilla, Venous malformation, Angiogenesis, Case report

Core tip: Hemophilic pseudotumor (HP) is a rare complication in patients with hemophilia. We present herein 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. This case highlights that the potential angiogenesis characterized by disturbed wound healing may lead to vascular malformation.