Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Dec 6, 2021; 9(34): 10681-10688
Published online Dec 6, 2021. doi: 10.12998/wjcc.v9.i34.10681
Intravascular papillary endothelial hyperplasia as a rare cause of cervicothoracic spinal cord compression: A case report
Hong-Lin Gu, Xiao-Qing Zheng, Shi-Qiang Zhan, Yun-Bing Chang
Hong-Lin Gu, Xiao-Qing Zheng, Shi-Qiang Zhan, Yun-Bing Chang, Department of Spine Surgery, Guangdong Provincial People’s Hospital, Guangdong Academy of Medical Sciences, Guangzhou 510080, Guangdong Province, China
Author contributions: All authors of this manuscript have actively participated in the data acquisition, and they all have read and approved the final version of the manuscript; Gu HL and Zheng XQ collected the clinical data and drafted the main manuscript text; Zhan SQ and Chang YB performed the surgery and revised the paper.
Supported by Guangdong Medical Science and Technology Research Fund Project, No. A2021454.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
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: Yunbing Chang, MD, Chief Doctor, Department of Spine Surgery, Guangdong Provincial People’s Hospital, Guangdong Academy of Medical Sciences, No. 106 Zhongshan Road II, Guangzhou 510080, Guangdong Province, China. sygkspine@163.com
Received: April 20, 2021
Peer-review started: April 20, 2021
First decision: June 23, 2021
Revised: June 28, 2021
Accepted: September 14, 2021
Article in press: September 14, 2021
Published online: December 6, 2021
Processing time: 223 Days and 15.7 Hours
Abstract
BACKGROUND

Intravascular papillary endothelial hyperplasia (IPEH) is a rare benign reactive vascular lesion that grows into an expansile compressing mass. It most commonly involves the skin and subcutaneous tissue. Spinal involvement is rare, with only 11 reported cases in the literature. We report, to our knowledge, the first case of IPEH in the cervicothoracic spinal canal and present a literature review.

CASE SUMMARY

A 27-year-old man presented with acute-onset neck pain, numbness, and weakness in his extremities. Magnetic resonance imaging showed an epidural mass in the cervicothoracic (C6-T1) spinal canal and vertebral hemangioma (VH) involving the C7 vertebral body. C6-T1 Laminectomy and radical excision of the mass were performed. Histopathological examinations revealed papillary proliferation of vascular endothelial cells with thrombus formation, and an IPEH diagnosis was made. By his 6-mo follow-up appointment, his symptoms were relieved without recurrence. The possible pathogenesis, clinical and imaging features, differential diagnosis, and management of IPEH were reviewed.

CONCLUSION

We report, to our knowledge, the first case of IPEH in the cervicothoracic spinal canal, treated via complete resection, and showing a favorable outcome. We found a causal relationship between spinal IPEH and VH; this partly explains the mechanism of IPEH.

Keywords: Intravascular papillary endothelial hyperplasia; Cervicothoracic; Spinal cord compression; Thrombosis; Hemangioma; Case report

Core Tip: Intravascular papillary endothelial hyperplasia (IPEH) is a rare benign reactive vascular lesion that grows into an expansile compressing mass. Spine involvement is rare, with only 11 case reports on its occurrence. We reported the first case of IPEH in the cervicothoracic spinal canal, which was treated via complete resection and had a good prognosis. We also found a causal relationship between spinal IPEH and vertebral hemangioma, and this partly explained the mechanism of IPEH.