Kim KW, Cho JH. Muscular atrophy and weakness in the lower extremities in Behçet’s disease: A case report and review of literature. World J Clin Cases 2021; 9(20): 5647-5654 [PMID: 34307620 DOI: 10.12998/wjcc.v9.i20.5647]
Corresponding Author of This Article
Jae-Heung Cho, PhD, Associate Professor, Doctor, Department of Korean Medicine Rehabilitation, College of Korean Medicine, Kyung Hee University, 26 Kyungheedae-ro, Dongdaemun-gu, Seoul 02447, South Korea. vetkong95@hanmail.net
Research Domain of This Article
Clinical Neurology
Article-Type of This Article
Case Report
Open-Access Policy of This Article
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/
World J Clin Cases. Jul 16, 2021; 9(20): 5647-5654 Published online Jul 16, 2021. doi: 10.12998/wjcc.v9.i20.5647
Muscular atrophy and weakness in the lower extremities in Behçet’s disease: A case report and review of literature
Koh-Woon Kim, Jae-Heung Cho
Koh-Woon Kim, Jae-Heung Cho, Department of Korean Medicine Rehabilitation, College of Korean Medicine, Kyung Hee University, Seoul 02447, South Korea
Author contributions: Kim KW was the patient’s attending physician; She reviewed the literature and contributed to manuscript drafting; Cho JH analyzed and interpreted the imaging findings and was responsible for the revision of the manuscript for important intellectual content; and all authors have read and approved the final manuscript.
Informed consent statement: Informed written consent was obtained from the patient for the publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflicts 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: Jae-Heung Cho, PhD, Associate Professor, Doctor, Department of Korean Medicine Rehabilitation, College of Korean Medicine, Kyung Hee University, 26 Kyungheedae-ro, Dongdaemun-gu, Seoul 02447, South Korea. vetkong95@hanmail.net
Received: March 4, 2021 Peer-review started: March 4, 2021 First decision: April 13, 2021 Revised: April 14, 2021 Accepted: May 20, 2021 Article in press: May 20, 2021 Published online: July 16, 2021 Processing time: 125 Days and 1.2 Hours
Abstract
BACKGROUND
In Behçet’s disease (BD), very few cases of muscular involvement have been reported previously. The natural history and therapeutic protocol for muscular involvement in BD are obscure due to the low incidence of peripheral neuropathy or myopathy in BD. The purpose of our study was to report a rare case of BD with chronic, focal forms of neuromyopathy and review the relevant literature.
CASE SUMMARY
We herein report the case of a 54-year-old man who presented with progressive muscular atrophy and weakness of both thighs 2 years after the presentation of the cardinal symptoms of BD. The past medical history, electrophysiological study, neurological examination, blood tests, magnetic resonance imaging study, and histological exam were performed for the differential diagnosis. Relevant literature on muscular involvement in BD was reviewed. Neurological examination revealed that muscular involvement was predominantly localized in the proximal parts of the lower extremities. Heterogeneous enhancement of several thigh muscles was observed on magnetic resonance imaging, which corresponded with the clinical manifestations. Histological study of one of the enhanced muscles showed denervation atrophy of the muscle with superimposed myopathic changes, while electrophysiological studies only suggested denervation.
CONCLUSION
To our knowledge, this is the first case of neurogenic muscular atrophy with a specific set of clinical, radiological, electrophysiological, and histological findings reported in BD.
Core Tip: Muscular involvement in Behçet’s disease (BD) has been rarely reported, along with the low incidence of peripheral neuropathy or myopathy in BD. Here, we present a rare case of BD with chronic, focal forms of neuromyopathy and review the relevant literature. To our knowledge, this is the first case of neurogenic muscular atrophy with a specific set of clinical, radiological, electrophysiological, and histological findings reported in BD. Two years after the presentation of the cardinal symptoms of BD, the patient presented with progressive muscular atrophy and weakness. This case highlights that despite the rare involvement of muscles, it should be considered, particularly in BD patients, that muscular symptoms with or without the cardinal symptoms of BD might be correlated with BD.
