Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Mar 6, 2021; 9(7): 1741-1747
Published online Mar 6, 2021. doi: 10.12998/wjcc.v9.i7.1741
Mononeuropathy multiplex associated with systemic vasculitis: A case report
Hyun Jun Chae, Jung Woo Kim, Yae Lim Lee, Jeong Hwan Park, Sang Yoon Lee
Hyun Jun Chae, Jung Woo Kim, Yae Lim Lee, Department of Rehabilitation Medicine, Seoul National University College of Medicine, Seoul National University Hospital, Seoul 07061, South Korea
Jeong Hwan Park, Department of Pathology, Seoul National University College of Medicine, SMG-SNU Boramae Medical Center, Seoul 07061, South Korea
Sang Yoon Lee, Department of Rehabilitation Medicine, Seoul National University College of Medicine, SMG-SNU Boramae Medical Center, Seoul 07061, South Korea
Author contributions: Chae HJ managed the patient and wrote the manuscript; Kim JW and Lee YL participated in data collection and revised the manuscript; Park JH provided histology images and interpretation; Lee SY was responsible for the clinical management of the patient, drafting and editing of the manuscript; all authors read and approved the final 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: 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: Sang Yoon Lee, MD, PhD, Associate Professor, Department of Rehabilitation Medicine, Seoul National University College of Medicine, SMG-SNU Boramae Medical Center, 20 Boramae-ro 5-gil, Dongjak-gu, Seoul 07061, South Korea. rehabilee@gmail.com
Received: November 18, 2020
Peer-review started: November 18, 2020
First decision: December 30, 2020
Revised: January 5, 2021
Accepted: January 21, 2021
Article in press: January 21, 2021
Published online: March 6, 2021
Processing time: 102 Days and 16.3 Hours
Abstract
BACKGROUND

Vasculitis, a systemic disorder with inflammation of blood vessel walls, can develop broad spectrum of signs and symptoms according to involvement of various organs, and therefore, early diagnosis of vasculitis is challenging. We herein describe a patient who developed a special case of systemic vasculitis with mononeuropathy multiplex, rectal perforation and antiphospholipid syndrome (APS) presented with pulmonary embolism.

CASE SUMMARY

A 61-year-old woman visited hospital with complaints of myalgia and occasional fever. She was initially diagnosed as proctitis and treated with antibiotics, however, there was no improvement. In addition, she also complained right foot drop with hypesthesia, and left 2nd and 3rd finger tingling sensation. She underwent nerve conduction study for evaluation, and it revealed sensorimotor polyneuropathy in the left arm and bilateral legs. Subsequent sural nerve biopsy strongly suggested vasculitic neuropathy. Based on nerve biopsy and clinical manifestation, she was diagnosed with vasculitis and treated with immuno-suppressive therapy. During treatment, sudden rectal perforation and pulmonary thromboembolism occurred, and further laboratory study suggested probable concomitant APS. Emergency Hartmann operation was performed for rectal perforation, and anti-coagulation therapy was started for APS. After few cycles of immunosuppressive therapy, tingling sensation and weakness in her hand and foot had been partially recovered and vasculitis was considered to be stationary.

CONCLUSION

Vasculitis can be presented with a variety of signs and symptoms, therefore, clinicians should always consider the possibility of diagnosis.

Keywords: Systemic Vasculitis; Peripheral nervous system diseases; Mononeuropathies; Antiphospholipid syndrome; Pulmonary embolism; Case report

Core Tip: We present a special case of systemic vasculitis with initially developed a mononeuropathy multiplex, followed by rectal perforation, and antiphospholipid syndrome presented with pulmonary embolism. Systemic vasculitis can be presented with a variety of signs and symptoms, therefore, clinicians should always consider the possibility of diagnosis.