Case Reports
24 September 2024
Vol. 76 No. 4 (2024)
Cervical myelopathy as an atypical presentation of antineutrophil cytoplasmic antibody-associated vasculitis in a patient affected by silicosis: a case report and literature overview
Publisher's note
All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article or claim that may be made by its manufacturer is not guaranteed or endorsed by the publisher.
All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article or claim that may be made by its manufacturer is not guaranteed or endorsed by the publisher.
750
Views
166
Downloads
Authors
We describe the case of a 76-year-old man affected by pneumoconiosis, secondary to silica dust exposure, who was diagnosed with antineutrophil cytoplasmic antibody (ANCA)-positive microscopic polyangiitis (MPA)-related cervical myelitis. Pneumoconiosis is reported to trigger autoantibody production and the onset of different autoimmune diseases, including ANCA-associated vasculitis (AAV). MPA is an AAV of the small vessels that can often affect the nervous system, although involvement of the spinal cord in the form of myelitis is described as an anecdotal occurrence. Our experience suggests that an autoimmunity workup should be considered for patients with pneumoconiosis who present with neurological symptoms consistent with AAV.
Altmetrics
Downloads
Download data is not yet available.
Citations
Gómez-Puerta JA, Gedmintas L, Costenbader KH. The association between silica exposure and development of ANCA-associated vasculitis: systematic review and meta-analysis. Autoimmun Rev 2013; 12: 1129-35. DOI: https://doi.org/10.1016/j.autrev.2013.06.016
Sassi SB, Ghorbel IB, Mizouni H, Houman MH, Hentati F. Microscopic polyangiitis presenting with peripheral and central neurological manifestations. Neurol Sci 2011; 32: 727-9. DOI: https://doi.org/10.1007/s10072-011-0653-x
Koike H, Nishi R, Ohyama K, Morozumi S, Kawagashira Y, Furukawa S, et al. ANCA-associated vasculitic neuropathies: a review. Neurol Ther 2022; 11: 21-38. DOI: https://doi.org/10.1007/s40120-021-00315-7
Shimojima Y, Kishida D, Ichikawa T, Kida T, Yajima N, Omura S, et al. Hypertrophic pachymeningitis in ANCA-associated vasculitis: a cross-sectional and multi-institutional study in Japan (J-CANVAS). Arthritis Res Ther 2022; 24: 204. DOI: https://doi.org/10.1186/s13075-022-02898-4
Shimojima Y, Sekijima Y. Hypertrophic pachymeningitis in ANCA-associated vasculitis: clinical and immunopathological features and insights. Autoimmun Rev 2023; 22: 103338. DOI: https://doi.org/10.1016/j.autrev.2023.103338
Saeki T, Fujita N, Kourakata H, Yamazaki H, Miyamura S. Two cases of hypertrophic pachymeningitis associated with myeloperoxidase antineutrophil cytoplasmic autoantibody (MPO-ANCA)-positive pulmonary silicosis in tunnel workers. Clin Rheumatol 2004; 23: 76-80. DOI: https://doi.org/10.1007/s10067-003-0815-1
Wang DC, Wei JW, Liu JH, Hu YG. The upper thoracic spinal cord compression as the initial manifestation of Wegener’s granulomatosis: a case report. Eur Spine J 2007; 16: 296-300. DOI: https://doi.org/10.1007/s00586-007-0318-x
Xie J, Jia E, Wang S, Yu Y, Li Z, Zhang J, et al. Relapsing subarachnoid hemorrhage as a clinical manifestation in microscopic polyangiitis: a case report and literature review. Clin Rheumatol 2022; 41: 3227-35. DOI: https://doi.org/10.1007/s10067-022-06163-6
Decker ML, Emery DJ, Smyth PS, Lu JQ, Lacson A, Yacyshyn E. Microscopic polyangiitis with spinal cord involvement: a case report and review of the literature. J Stroke Cerebrovasc Dis 2016; 25: 1696-704. DOI: https://doi.org/10.1016/j.jstrokecerebrovasdis.2016.01.034
Long Y, Zheng Y, Chen M, Zhang B, Gao C, Gao Q, et al. Antineutrophil cytoplasmic antibodies in patients with idiopathic inflammatory-demyelinating diseases. Neuroimmunomodulation 2014; 21: 297-303. DOI: https://doi.org/10.1159/000357681
Martens J. Spinal cord involvement in Wegener’s granulomatosis. Clin Rheumatol 1982; 1: 221. DOI: https://doi.org/10.1007/BF02042779
Reinhold-Keller E, de Groot K, Holl-Ulrich K, Arlt AC, Heller M, Feller AC, et al. Severe CNS manifestations as the clinical hallmark in generalized Wegener’s granulomatosis consistently negative for antineutrophil cytoplasmic antibodies (ANCA). A report of 3 cases and a review of the literature. Clin Exp Rheumatol 2001; 19: 541-9
Hamilton AJ, Whitehead DJ, Bull MD, D’Souza RJ. Systemic panca-associated vasculitis with central nervous involvement causing recurrent myelitis: case report. BMC Neurol 2010; 10: 118. DOI: https://doi.org/10.1186/1471-2377-10-118
Weidner S, Hafezi-Rachti S, Rupprecht HD. Thromboembolic events as a complication of antineutrophil cytoplasmic antibody-associated vasculitis. Arthritis Care Res 2006; 55: 146-9. DOI: https://doi.org/10.1002/art.21704
How to Cite
Cervical myelopathy as an atypical presentation of antineutrophil cytoplasmic antibody-associated vasculitis in a patient affected by silicosis: a case report and literature overview. (2024). Reumatismo, 76(4). https://doi.org/10.4081/reumatismo.2024.1702
Copyright (c) 2024 the Author(s)
This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.
PAGEPress has chosen to apply the Creative Commons Attribution NonCommercial 4.0 International License (CC BY-NC 4.0) to all manuscripts to be published.
