Case Report
Copyright ©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jan 26, 2020; 8(2): 318-324
Published online Jan 26, 2020. doi: 10.12998/wjcc.v8.i2.318
Cervicogenic exophthalmos: Possible etiology and pathogenesis
Chi-Ming Wu, Hung-En Liao, Shang-Wei Hsu, Shou-Jen Lan
Chi-Ming Wu, Orthopaedic Department, Jen-Ai Hospital, Taichung 40041, Taiwan
Chi-Ming Wu, Orthopaedic Department, National Defense Medical Center, Taipei 11465, Taiwan
Chi-Ming Wu, Hung-En Liao, Shang-Wei Hsu, Shou-Jen Lan, Department of Healthcare Administration, Asia University, Taichung 41354, Taiwan
Shou-Jen Lan, Department of Medical Research, China Medical University Hospital, China Medical University, Taichung 40447, Taiwan
Author contributions: Wu CM treated the patient and wrote the manuscript; Lan SJ analysed the patient’s clinical data and submitted the manuscript; Liao HE and Hsu SW were involved in the concept and revised the manuscript; all authors approved the final version of this manuscript.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: All authors, including Chi-Ming Wu, Hung-En Liao, Shang-Wei Hsu and Shou-Jen Lan, all 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 which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (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: Shou-Jen Lan, PhD, Professor, Department of Healthcare Administration, Asia University, No. 500 Lioufeng Road, Wufeng District, Taichung City 41354, Taiwan. shoujenlan@gmail.com
Received: October 2, 2019
Peer-review started: October 2, 2019
First decision: December 4, 2019
Revised: December 17, 2019
Accepted: December 22, 2019
Article in press: December 22, 2019
Published online: January 26, 2020
Processing time: 106 Days and 13.2 Hours
Abstract
BACKGROUND

Unilateral exophthalmos is often caused by inflammation, neoplasm, infection, metabolic disease, vascular disorder and several other less common conditions. Reflex sympathetic dystrophy related to unilateral exophthalmos has not been reported in the past literature.

CASE SUMMARY

We describe a 45-year-old female with unilateral exophthalmos caused by reflex sympathetic dystrophy and its unexpected spontaneous disappearance after a standard anterior cervical discectomy and fixation operation with two PEEK interbody cages and a plate. To our surprise, the patient’s left unilateral exophthalmos improved spontaneously in the morning on postoperative day 2-with no relapse, without any further medication, as of seven years. We have named this condition “cervicogenic exophthalmos.”

CONCLUSION

We would inform other clinicians that unilateral exophthalmos was caused not only by inflammation, vascular disorder, infection, neoplasm, or metabolic disease, but also by reflex sympathetic dystrophy related with cervicogenic spondylosis. To the best of our knowledge, ours is the first related case report and use of the term “cervicogenic exophthalmos” after reviewing previous literature.

Keywords: Exophthalmos; Reflex sympathetic dystrophy; Anterior cervical discectomy and fixation; PEEK cage; Cervicogenic exophthalmos; Cervicogenic spondylosis; Case report

Core tip: “Cervicogenic” headache, vertigo, dizziness, hypertension, or tinnitus had been reported that patients with cervical spondylosis caused by sympathetic symptoms including headache, vertigo, dizziness, hypertension, or tinnitus and treated successfully with operation of anterior cervical discectomy and fixation using PEEK cages and a plate. Here we described a patient with spondylosis of C4/5/6 with unilateral exophthalmos. She suffered from unexpected spontaneous improvement of unilateral exophthalmos after an operation of anterior cervical discectomy and fixation with two PEEK interbody cages and a plate. Therefore, we have named the condition “cervicogenic exophthalmos”. To the best of our knowledge, ours is the first related case report and use of the term “cervicogenic exophthalmos” after reviewing previous literature.