Macrophagic myofasciitis: an atypical presentation for a rare disease with a challenging approach
More details
Hide details
Internal Medicine Department, Oporto University Hospital Center, Portugal
Clinical Immunology Unit, Oporto University Hospital Center, Portugal
Unit for Multidisciplinary Research in Biomedicine (UMIB) – ICBAS, Oporto University, Portugal
Abel Salazar Institute of Biomedical Sciences (ICBAS), Oporto University, Portugal
Submission date: 2020-02-21
Final revision date: 2020-05-23
Acceptance date: 2020-06-02
Online publication date: 2020-06-29
Publication date: 2020-06-29
Reumatologia 2020;58(3):167-172
Macrophagic myofasciitis (MMF) is a rare immune-mediated myopathy that seems to be triggered by aluminium hydroxide adjuvant used in vaccines. Its presentation is relatively heterogeneous and treatment with steroids leads to improvement, although there is little evidence regarding the role of other immunosuppressants. The histological findings in MMF seem to be the result of an abnormal presence in the inoculation site of aluminium, which can induce an immune-mediated muscular disease in susceptible persons. The authors describe the case of a patient with an atypical presentation of macrophagic myofasciitis, with histological confirmation in a muscle biopsy distant from the inoculation site, and a good therapeutic response to tacrolimus and mycophenolate mofetil, as well as a discussion on the pathologic basis, controversies and emerging treatments for this condition.
Gherardi RK, Authier FJ. Macrophagic myofasciitis: characterization and pathophysiology. Lupus 2012; 21: 184-189, DOI: 10.1177/0961203311429557.
Israeli E, Agmon-Levin N, Blank M, Shoenfeld Y. Macrophagic myofaciitis a vaccine (alum) autoimmune-related disease. Clin Rev Allergy Immunol 2011; 41: 163-168, DOI: 10.1007/s12016-010-8212-4.
Gherardi RK, Coquet M, Cherin P, et al. Macrophagic myofasciitis lesions assess long-term persistence of vaccine – derived aluminium hydroxide in muscle. Brain 2001; 124: 1821-1831, DOI: 10.1093/brain/124.9.1821.
Chkheidze R, Burns DK, White CL, et al. Morin Stain Detects Aluminum-Containing Macrophages in Macrophagic Myofasciitis and Vaccination Granuloma With High Sensitivity and Specificity. J Neuropathol Exp Neurol 2017; 76: 323-331, DOI: 10.1093/jnen/nlx011.
Perricone C, Colafrancesco S, Mazor RD, et al. Autoimmune/inflammatory syndrome induced by adjuvants (ASIA) 2013: Unveiling the pathogenic, clinical and diagnostic aspects.
J Autoimmun 2013; 47: 1-16, DOI: 10.1016/j.jaut.2013.10.004.
Shoenfeld Y, Agmon-Levin N. ‘ASIA’ – autoimmune/inflammatory syndrome induced by adjuvants. J Autoimmun 2011; 36: 4-8, DOI: 10.1016/j.jaut.2010.07.003.
Gherardi RK, Crépeaux G, Authier FJ. Myalgia and chronic fatigue syndrome following immunization: macrophagic myofasciitis and animal studies support linkage to aluminum adjuvant persistency and diffusion in the immune system. Autoimmun Rev 2019; 18: 691-705, DOI: 10.1016/j.autrev.2019.05.006.
Awate S, Babiuk LA, Mutwiri G. Mechanisms of action of adjuvants. Front Immunol 2013; 4: 114, DOI: 10.3389/fimmu.2013.00114.
McKee AS, MacLeod MKL, Kappler JW, Marrack P. Immune mechanisms of protection: can adjuvants rise to the challenge? BMC Biol 2010; 8: 37, DOI: 10.1186/1741-7007-8-37.
Exley C, Siesjö P, Eriksson H. The immunobiology of aluminium adjuvants: how do they really work? Trends Immunol 2010; 31: 103-109, DOI: 10.1016/j.it.2009.12.009.
Kumagai Y, Shiokawa Y, Medsger TA Jr., Rodnan GP. Clinical spectrum of connective tissue disease after cosmetic surgery. Observations on eighteen patients and a review of the Japanese literature. Arthritis Rheum 1984; 27: 1-12, DOI: 10.1002/art.1780270101.
Sergott TJ, Limoli JP, Baldwin CM Jr., Laub DR. Human adjuvant disease, possible autoimmune disease after silicone implantation: a review of the literature, case studies, and speculation for the future. Plast Reconstr Surg 1986; 78: 104-114, DOI: 10.1097/00006534-198607000-00018.
Satoh M, Kuroda Y, Yoshida H, et al. Induction of lupus autoantibodies by adjuvants. J Autoimmun 2003; 21: 1-9, DOI: 10.1016/s0896-8411(03)00083-0.
Kuroda Y, Nacionales DC, Akaogi J, et al. Autoimmunity induced by adjuvant hydrocarbon oil components of vaccine. Biomed Pharmacother 2004; 58: 325-337, DOI: 10.1016/j.biopha.2004.04.009.
Agmon-Levin N, Shoenfeld Y. Chronic fatigue syndrome with autoantibodies –the result of an augmented adjuvant effect of hepatitis-B vaccine and silicone implant. Autoimmun Rev 2008; 8: 52-55, DOI: 10.1016/j.autrev.2008.07.026.
Burek CL, Talor MV. Environmental triggers of autoimmune thyroiditis. J Autoimmun 2009; 33: 183-189, DOI: 10.1016/j.jaut.2009.09.001.
Arango MT, Perricone C, Kivity S, et al. HLA-DRB1 the notorious gene in the mosaic of autoimmunity. Immunol Res 2017; 65: 82-98, DOI: 10.1007/s12026-016-8817-7.
Guis S, Mattei JP, Nicoli F, et al. Identical twins with macrophagic myofasciitis: genetic susceptibility and triggering by aluminic vaccine adjuvants? Arthritis Rheum 2002; 47: 543-545, DOI: 10.1002/art.10666.
WHO. Macrophagic myofasciitis and aluminium-containing vaccines. WHO Weekly Epidemiological Record 1999.
WHO. Questions and Answers about macrophagic myofasciitis (MMF), 2008.
Goullé JP, Grangeot-Keros L. Aluminum and vaccines: Current state of knowledge. Med Mal Infect 2020; 50: 16-21, DOI: 10.1016/j.medmal.2019.09.012.
Segal Y, Dahan S, Sharif K, et al. The value of Autoimmune Syndrome Induced by Adjuvant (ASIA) – Shedding light on orphan diseases in autoimmunity. Autoimmun Rev 2018; 17: 440-448, DOI: 10.1016/j.autrev.2017.11.037.
Ameratunga R, Gillis D, Gold M, et al. Evidence Refuting the Existence of Autoimmune/Autoinflammatory Syndrome Induced by Adjuvants (ASIA). J Allergy Clin Immunol Pract 2017; 5: 1551-1555.e1, DOI: 10.1016/j.jaip.2017.06.033.
Liang R, Liu A, Perumpail RB, et al. Advances in alcoholic liver disease: An update on alcoholic hepatitis. World J Gastroenterol 2015; 21: 11893-11903, DOI: 10.3748/wjg.v21.i42.11893.
Authier FJ, Sauvat S, Champey J, et al. Chronic fatigue syndrome in patients with macrophagic myofasciitis. Arthritis Rheum 2003; 48: 569-570, DOI: 10.1002/art.10740.
Butnaru D, Shoenfeld Y. Adjuvants and lymphoma risk as part of the ASIA spectrum. Immunol Res 2015; 61: 79-89, DOI: 10.1007/s12026-014-8622-0.
Gherardi RK, Coquet M, Chérin P, et al. Macrophagic myofasciitis: an emerging entity. Groupe d’Etudes et Recherche sur les Maladies Musculaires Acquises et Dysimmunitaires (GERMMAD) de l’Association Française contre les Myopathies (AFM). Lancet 1998; 352: 347-352, DOI: 10.1016/s0140-6736(98)02326-5.
Exley C, Swarbrick L, Gherardi RK, Authier FJ. A role for the body burden of aluminium in vaccine-associated macrophagic myofasciitis and chronic fatigue syndrome. Med Hypotheses 2009; 72: 135-139, DOI: 10.1016/j.mehy.2008.09.040.
Authier FJ, Chérin P, Creange A, et al. Central nervous system disease in patients with macrophagic myofasciitis. Brain 2001; 124: 974-983, DOI: 10.1093/brain/124.5.974.
Gherardi RK, Authier FJ. Aluminum inclusion macrophagic myofasciitis: a recently identified condition. Immunol Allergy Clin North Am 2003; 23: 699-712, DOI: 10.1016/s0889-8561(03)00095-x.
Ryan AM, Bermingham N, Harrington HJ, Keohane C. Atypical presentation of macrophagic myofasciitis 10 years post vaccination. Neuromuscul Disord 2006; 16: 867-869, DOI: 10.1016/j.nmd.2006.07.017.
Santiago T, Rebelo O, Negrão L, Matos A. Macrophagic myofasciitis and vaccination: consequence or coincidence? Rheumatol Int 2015; 35: 189-192, DOI: 10.1007/s00296-014-3065-4.
Rigolet M, Aouizerate J, Couette M, et al. Clinical features in patients with long-lasting macrophagic myofasciitis. Front Neurol 2014; 5: 230, DOI: 10.3389/fneur.2014.00230.
Olivo Pallo PA, de Souza FHC, Miossi R, Shinjo SK. Mycophenolate mofetil in patients with refractory systemic autoimmune myopathies: case series. Adv Rheumatol 2018; 58: 34, DOI: 10.1186/s42358-018-0035-7.
Hanaoka H, Iida H, Kiyokawa T, et al. Mycophenolate mofetil treatment with or without a calcineurin inhibitor in resistant inflammatory myopathy. Clin Rheumatol 2019; 38: 585-590, DOI: 10.1007/s10067-018-4294-9.
Copyright: © Narodowy Instytut Geriatrii, Reumatologii i Rehabilitacji w Warszawie. This is an Open Access journal, all articles are distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International (CC BY-NC-SA 4.0) License (https://creativecommons.org/licenses/by-nc-sa/4.0/), allowing third parties to copy and redistribute the material in any medium or format and to remix, transform, and build upon the material, provided the original work is properly cited and states its license.
Journals System - logo
Scroll to top