Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Dec 16, 2021; 9(35): 11024-11028
Published online Dec 16, 2021. doi: 10.12998/wjcc.v9.i35.11024
Severe absence of intra-orbital fat in a patient with orbital venous malformation: A case report
Lu-Di Yang, Shi-Qiong Xu, Ye-Fei Wang, Ren-Bing Jia
Lu-Di Yang, Shi-Qiong Xu, Ye-Fei Wang, Ren-Bing Jia, Department of Ophthalmology, Ninth People’s Hospital, Shanghai JiaoTong University School of Medicine, Shanghai 200025, China
Lu-Di Yang, Shanghai Key Laboratory of Orbital Diseases and Ocular Oncology, Shanghai 200025, China
Author contributions: Yang LD drafted the manuscript; Yang LD, Xu SQ and Wang YF performed the case and collected the images; Jia RB reviewed the manuscript and offered clinical advice.
Supported by the National Natural Science Foundation of China, No. 81770961.
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 competing interests.
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: Ren-Bing Jia, MD, PhD, Doctor, Professor, Department of Ophthalmology, Ninth People’s Hospital, Shanghai JiaoTong University School of Medicine, No. 639 Zhizaoju Road, Shanghai 200025, China. renbingjia@sjtu.edu.cn
Received: May 23, 2021
Peer-review started: May 23, 2021
First decision: July 15, 2021
Revised: July 26, 2021
Accepted: October 25, 2021
Article in press: October 25, 2021
Published online: December 16, 2021
Processing time: 200 Days and 15.6 Hours
Abstract
BACKGROUND

The orbital venous malformation is quite common in orbital diseases. Clinically, it is usually characterized by proptosis. However, among patients with distensible venous malformations, if the lesions continuously progress, they may induce enlargement of the orbital bone or orbital lipoatrophy, which in turn leads to enophthalmos.

CASE SUMMARY

Here, we report a patient who presented with enophthalmos and had a severe absence of intra-orbital fat secondary to orbital venous malformation. The patient was a 66-year-old female with a 20-year history of enophthalmos. Hertel exophthalmometry readings in a relaxed upright position were 4 mm OD and 13 mm OS with a 97 mm base. It was determined that she had positional “proptosis”. Physical examination also revealed a bulging mass on her hard palate. Computed tomographic scan and magnetic resonance imaging showed an expansion of the right orbit with local bony defects and multiple soft-tissue masses.

CONCLUSION

Long-term lack of awareness about the presence of orbital venous malformations, persistent venous congestion could lead to compression of the orbital fat, which in turn induces atrophy or the absence of intra-orbital fat.

Keywords: Absence of intra-orbital fat; Venous malformation; Enophthalmos; Entropion; Long medical history; Case report

Core Tip: Timely diagnosis and treatment are critical to prevent secondary irreversible lesions. In patients with severe orbital venous malformations, delayed diagnosis and treatment may induce atrophy or the absence of intra-orbital fat. This case report provides a cautionary tale for patients with a long history of orbital venous malformations.