Observational Study
Copyright ©The Author(s) 2019. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jun 6, 2019; 7(11): 1282-1290
Published online Jun 6, 2019. doi: 10.12998/wjcc.v7.i11.1282
Clinical features of syphilitic myelitis with longitudinally extensive myelopathy on spinal magnetic resonance imaging
Jun-Liang Yuan, Wei-Xue Wang, Wen-Li Hu
Jun-Liang Yuan, Wen-Li Hu, Department of Neurology, Beijing Chaoyang Hospital, Capital Medical University, Beijing 100020, China
Wei-Xue Wang, Department of Oncology, Beijing Chaoyang Hospital, Capital Medical University, Beijing 100020, China
Author contributions: Yuan JL and Wang WX are co-first authors; Yuan JL, Wang WX, and Hu WL designed and performed the research; Yuan JL and Wang WX collected and analyzed the data; Yuan JL and Wang WX wrote the paper.
Supported by the National Natural Science Foundation of China, No. 81301016; and the Beijing Municipal Administration of Hospitals Incubating Program, No. PX2019009.
Institutional review board statement: Our work was approved by the Ethics Committee of Beijing Chaoyang Hospital, Capital Medical University.
Informed consent statement: The patient gave informed consent.
Conflict-of-interest statement: The authors declare that they have no competing interests.
Data sharing statement: No additional data are available.
STROBE statement: The authors have read the STROBE Statement–checklist of items, and the manuscript was prepared and revised according to the STROBE Statement–checklist of items.
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: Wen-Li Hu, MD, Director, Doctor, Department of Neurology, Beijing Chaoyang Hospital, Capital Medical University, 8 Gongti South, Chaoyang District, Beijing 100020, China. wenlihu3366@126.com
Telephone: +86-10-85231376 Fax: +86-10-85231376
Received: January 23, 2019
Peer-review started: January 23, 2019
First decision: March 14, 2019
Revised: April 16, 2019
Accepted: May 2, 2019
Article in press: May 2, 2019
Published online: June 6, 2019
Processing time: 135 Days and 14.7 Hours
Abstract
BACKGROUND

Syphilitic myelitis caused by Treponema pallidum is an extremely rare disease. However, symptomatic neurosyphilis, especially syphilitic myelitis, and its clinical features have been infrequently reported. Only a few cases of syphilitic myelitis have been documented. To the best of our knowledge, there are only 19 reported cases of syphilitic myelitis. However, the clinical features of syphilitic myelitis with longitudinally extensive myelopathy have been still not clear.

AIM

To explore the clinical features of syphilitic myelitis with longitudinally extensive myelopathy on spinal magnetic resonance imaging (MRI).

METHODS

First, we report a patient who suffered from syphilitic myelitis with symptoms of sensory disturbance, with longitudinally extensive myelopathy with "flip-flop sign" on spinal MRI. Second, we performed a literature search to identify other reports (reviews, case reports, or case series) from January 1987 to December 2018, using the PubMed and Web of Science databases with the terms including "syphilis", "neurosyphilis", "syphilitic myelitis", "meningomyelitis", "central nervous system", and "spine". We also summarized the clinical features of syphilitic myelitis with longitudinally extensive myelopathy.

RESULTS

A total of 16 articles of 20 cases were identified. Sixteen patients presented with the onset of sensory disturbance (80%), 15 with paraparesis (75%), and 9 with urinary retention (45%). Eleven patients had a high risk behavior (55%). Five patients had concomitant human immunodeficiency virus infection (25%). Serological data showed that 15 patients had positive venereal disease research laboratory test (VDRL)/treponema pallidum particle agglutination (TPHA), and 17 had positive VDRL/TPHA in cerebrospinal fluid (CSF). Seventeen patients were found to have elevated leukocytosis and protein in CSF. On MRI, 16 patients showed abnormal hyperintensities involved the thoracic spine, 6 involved the cervical spine, and 3 involved both the cervical and thoracic spine. There were 3 patients with the "flip-flop sign". All the patients were treated with penicillin, and 15 patients had a good prognosis.

CONCLUSION

Our case further raises awareness of syphilitic myelitis as an important complication of neurosyphilis due to homosexuality, especially in developing countries such as China.

Keywords: Neurosyphilis; Syphilitic myelitis; Syphilitic meningomyelitis; Human immunodeficiency virus

Core tip: Syphilitic myelitis is a very rare manifestation of neurosyphilis. Early diagnosis and treatment are crucial because it represents a treatable and potentially reversible cause of myelopathy if treated with penicillin. Herein, we report a 25-year-old young man presenting with symptoms of sensory disturbance, due to syphilitic myelitis with longitudinally extensive myelopathy with "flip-flop sign" on spinal magnetic resonance imaging. Furthermore, we summarized the clinical features of syphilitic myelitis with longitudinally extensive myelopathy by reviewing the relevant literature. Our study also raises awareness of an important complication of neurosyphilis due to homosexuality. Attention is drawn upon the importance of doing serological tests for syphilis when any atypical neurological disorders are presented.