Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. Feb 21, 2022; 28(7): 755-762
Published online Feb 21, 2022. doi: 10.3748/wjg.v28.i7.755
Crohn’s disease-related ‘gastrocnemius myalgia syndrome’ successfully treated with infliximab: A case report
Julien Catherine, Hazim Kadhim, Frédéric Lambot, Claire Liefferinckx, Virginie Meurant, Lukas Otero Sanchez
Julien Catherine, Institute for Medical Immunology, Université Libre de Bruxelles, Gosselies 6041, Belgium
Julien Catherine, Department of Internal Medicine, C.U.B. Hôpital Erasme, Brussels 1070, Belgium
Hazim Kadhim, NeuroMyopathology Unit (Anatomic Pathology Service) and Reference Center for Neuromuscular Pathology, CHU Brugmann, Université Libre de Bruxelles, Brussels 1020, Belgium
Frédéric Lambot, Department of Internal Medicine, Centre Hospitalier Universitaire Tivoli, La Louvière 7100, Belgium
Claire Liefferinckx, Lukas Otero Sanchez, Department of Gastroenterology, Hepatopancreatology and Digestive Oncology, C.U.B. Hôpital Erasme, Brussels 1070, Belgium
Claire Liefferinckx, Lukas Otero Sanchez, Laboratory of Experimental Gastroenterology, Université Libre de Bruxelles, Brussels 1070, Belgium
Virginie Meurant, Department of Emergency Medicine, Centre Hospitalier Universitaire Tivoli, La Louvière 7100, Belgium
Author contributions: Lambot F, Catherine J and Otero Sanchez L managed the patient; Kadhim H did myopathology diagnosis and analyses; Catherine J, Otero Sanchez L and Meurant V designed the study and collected data; Catherine J and Otero Sanchez L drafted and wrote the manuscript; Liefferinckx C, Lambot F and Kadhim H provided their expertise throughout the course of the work by revising and editing the manuscript; all authors approved the final version of the manuscript to be published.
Supported by National Fund for Scientific Research (F.R.S-FNRS) as research fellows to Catherine J and Otero Sanchez L; Fonds Erasme to Otero Sanchez L; and F.R.S-FNRS as postdoctoral fellow to Liefferinckx C.
Informed consent statement: The study was performed in accordance with the ethical standards of the Helsinki Declaration and was approved by the institutional review board of CHU Tivoli (approval number #1396). Patient’s informed consent was obtained by Meurant V.
Conflict-of-interest statement: Liefferinckx C received consultancy fees from Takeda and Galapagos; speaker fees from Sandoz, Janssen and AbbVie.
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: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Julien Catherine, MD, Medical Assistant, Research Fellow, Institute for Medical Immunology, Université Libre de Bruxelles, Rue Adrienne Boland 8, Gosselies 6041, Belgium. julien.catherine@ulb.be
Received: September 30, 2021
Peer-review started: September 30, 2021
First decision: December 4, 2021
Revised: December 13, 2021
Accepted: January 19, 2022
Article in press: January 19, 2022
Published online: February 21, 2022
Abstract
BACKGROUND

Extra-intestinal manifestations in inflammatory bowel diseases (IBD) are frequent and involve virtually all organs. Conversely, the clinical characteristics and course of inflammatory myopathies in IBD remain poorly described and mostly related to orbital myositis. Moreover, alternative therapeutic strategies in non-responder patients to corticosteroid therapy must still be clarified.

CASE SUMMARY

A 33-year-old woman with a history of unclassified colitis presented with acute bilateral calf pain. On admission, her clinical and biological examinations were non-specific. However, magnetic resonance imaging showed bilateral inflammatory changes in gastrocnemius muscles suggestive of myositis. Muscle biopsy confirmed the diagnosis of myositis and demonstrated an inflammatory infiltrate mainly located in the perimysial compartment including lympho-plasmocytic cells with the formation of several granulomatous structures while the endomysium was relatively spared. The combined clinical, biological and histomyopathological findings were concordant with the diagnosis of ‘gastrocnemius myalgia syndrome’ (GMS), a rare disorder associated with Crohn’s disease (CD). Ileocolonoscopy confirmed CD diagnosis and systemic corticosteroids (CS) therapy was started, resulting in a rapid clinical improvement. During CS tapering, however, she experienced a relapse of GMS together with a severe active ileocolitis. Infliximab was started and allowed a sustained remission of both conditions at the latest follow-up (20 mo).

CONCLUSION

The GMS represent a rare CD-associated inflammatory myopathy for which anti-tumour necrosis factor-α therapy might be considered as an effective therapeutic option.

Keywords: Crohn’s disease, Extra-intestinal manifestation, Gastrocnemius myalgia syndrome, Granulomatous myositis, Anti-tumour necrosis factor-α therapy, Case report

Core Tip: Inflammatory myopathies are scarce in the setting of inflammatory bowel diseases (IBD) and could be wrongly attributed to IBD-related osteoarticular manifestations or to medications’ side effects. This case describes a very atypical presentation of myositis restricted to the legs called the ‘gastrocnemius myalgia syndrome’, an entity only described during Crohn’s disease. Its restricted location to the legs as well as normal creatine kinase levels in serum reflecting a predominant interstitial immune reaction are key characteristics in reported cases. Although corticosteroids are often used as first-line therapy, cortico-dependence is not rare and anti-tumour necrosis factor-α agents might represent an effective therapeutic option.