Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. May 6, 2022; 10(13): 4137-4144
Published online May 6, 2022. doi: 10.12998/wjcc.v10.i13.4137
Large vessel vasculitis with rare presentation of acute rhabdomyolysis: A case report and review of literature
Lan-Jun Fu, Shou-Ci Hu, Wen Zhang, Li-Qing Ye, Hong-Bo Chen, Xiao-Jun Xiang
Lan-Jun Fu, Shou-Ci Hu, Wen Zhang, Li-Qing Ye, Hong-Bo Chen, Xiao-Jun Xiang, Department of Nephrology, The First Affiliated Hospital of Zhejiang Chinese Medical University (Zhejiang Provincial Hospital of Traditional Chinese Medicine), Hangzhou 310006, Zhejiang Province, China
Author contributions: Fu LJ, Zhang W, and Chen HB reviewed the literature; Fu LJ drafted the manuscript; Hu SC performed the analysis and interpretation of data; Zhang W helped design the figures; Ye LQ helped with acquisition of data; Hong-Bo Chen HB helped with the manuscript revision; Xiang XJ helped with the conception and design of the study; all authors granted approval for the final submitted version.
Supported by Medical Health Science and Technology Project of Zhejiang Provincial Health Commission, No. 2021KY833; and Traditional Chinese Medical Science and Technology Project of Zhejiang Province, No. 2021ZB110.
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 have no conflicts of interest to declare.
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: Xiao-Jun Xiang, MBBS, Associate Chief Physician, Department of Nephrology, The First Affiliated Hospital of Zhejiang Chinese Medical University (Zhejiang Provincial Hospital of Traditional Chinese Medicine), No. 54 Youdian Road, Hangzhou 310006, Zhejiang Province, China. 309454905@qq.com
Received: June 9, 2021
Peer-review started: June 9, 2021
First decision: September 1, 2021
Revised: September 12, 2021
Accepted: March 16, 2022
Article in press: March 16, 2022
Published online: May 6, 2022
Abstract
BACKGROUND

Musculoskeletal involvement in primary large vessel vasculitis (LVV), including giant cell arteritis and Takayasu's arteritis (TAK), tends to be subacute. With the progression of arterial disease, patients may develop polyarthralgia and myalgias, mainly involving muscle stiffness, limb/jaw claudication, cold/swelling extremities, etc. Acute development of rhabdomyolysis in addition to aortic aneurysm is uncommon in LVV. Herein, we report a rare case of LVV with the first presentation of acute rhabdomyolysis.

CASE SUMMARY

A 70-year-old Asian woman suffering from long-term low back pain was hospitalized due to limb claudication, dark urine and an elevated creatine kinase (CK) level. After treatment with fluid resuscitation and antibiotics, the patient remained febrile. Her workup showed persistent elevated levels of inflammatory markers, and imaging studies revealed an aortic aneurysm. A decreasing CK was evidently combined with elevated inflammatory markers and negativity for anti-neutrophilic cytoplasmic antibodies. LVV was suspected and confirmed by magnetic resonance angiography and positron emission tomography with 18F-fluorodeoxyglucose/computed tomography. With a favourable response to immunosuppressive treatment, her symptoms resolved, and clinical remission was achieved one month later. However, after failing to follow the tapering schedule, the patient was readministered 25 mg/d prednisolone due to disease relapse. Follow-up examinations showed decreased inflammatory markers and substantial improvement in artery lesions after 6 mo of treatment. At the twelve-month follow-up, she was clinically stable and maintained on corticosteroid therapy.

CONCLUSION

An exceptional presentation of LVV with acute rhabdomyolysis is described in this case, which exhibited a good response to immunosuppressive therapy, suggesting consideration for a differential diagnosis when evaluating febrile patients with myalgia and elevated CK. Timely use of high-dose steroids until a diagnosis is established may yield a favourable outcome.

Keywords: Large vessel vasculitis, Rhabdomyolysis, Giant cell arteritis, 18F-fluorodeoxyglucose-positron emission tomography/computed tomography, Immunosuppressive treatment, Case report

Core Tip: Musculoskeletal involvement in large vessel vasculitis (LVV) usually manifests as polyarthralgia and myalgias but without elevated creatine kinase (CK) levels. Herein, we report a rare case of LVV with the first presentation of acute rhabdomyolysis. With the use of magnetic resonance angiography and 18F-fluorodeoxyglucose-positron emission tomography/computed tomography, LVV was properly diagnosed. Ultimately, the patient achieved a favourable outcome after timely and persistent use of glucocorticoids. We present a case to supply clues for unusual diagnoses of LVV for febrile patients with myalgia and elevated CK.