Journal List > J Rheum Dis > v.19(5) > 1063991

TOOLS
Han, Park, Ryu, Choi, Park, Lee, Song, Jung, and Lee: A Case of Macrophagic Myofasciitis Initially Thought to Be a Metastatic Infection
Case Report
Journal of Rheumatic Diseases

Journal of Rheumatic Diseases 2012; 19(5): 285-289.

Published online: 31 October 2012

DOI: https://doi.org/10.4078/jrd.2012.19.5.285

A Case of Macrophagic Myofasciitis Initially Thought to Be a Metastatic Infection

Miyeun Han1, Junwon Park1, Hanhee Ryu1, In Ah Choi1, Wanbeom Park1, Eun Bong Lee1, Yeong Wook Song1, Haeyoen Jung2, Eun Young Lee1

1Department of Internal Medicine, College of Medicine, Seoul National University, Seoul, Korea.

2Department of Pathology, College of Medicine, Seoul National University, Seoul, Korea.

Corresponding to: Eun Young Lee, Department of Internal Medicine, College of Meidicine, Seoul National University, 101, Yeongeon-dong, Jongno-gu, Seoul 110-744, Korea. elee@snu.ac.kr

Received 19 September 2011       Revised 25 October 2011       Accepted 25 October 2011

Copyright © 2012 by The Korean College of Rheumatology

(free-access):

This is a Free Access article, which permits unrestricted non-commerical use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

Macrophagic myofasciitis (MMF) is a rare disease, often associated with the pathological persistence of aluminum hydroxide used in some vaccines, and is characterized by macrophage infiltration of the muscle. We report a case of MMF, initially thought to be a metastatic infection. A 38-year-old woman presented with fever, as well as pain and weakness in both thighs. On physical examination both thighs were swollen and lower-extremity motor-power was decreased to grade III. Laboratory tests showed leukocytosis and elevation of acute phase reactants, but all muscle enzymes except lactate dehydrogenase (LDH) were within normal range. Initially metastatic infection was suspected but she was diagnosed with MMF by muscle biopsy showing heavy CD68 positive macrophage infiltration. Her myalgia and muscle weakness improved after systemic steroid treatment. This case suggests that MMF might be considered for a patient with unexplained inflammatory myopathy with or without a history of vaccination.

Keywords: Macrophagic myofasciitis, Vaccine, Steroid

Figures and Tables

Figure 1
(A) MRI Rt Thigh AXL FSE T2 and (B) MRI Hip AXL FSE T2 gadolinium enhanced show multifocal peripheral rim enhancing lesions (arrow) with perilesional edema involving (A) the right vastus lateralis, (B) bilateral gluteus muscles and intermedius muscles.
jrd-19-285-g001
Figure 2
Gun biopsy at right gluteus medius specimen shows severe atrophy, degeneration and regeneration of muscle fibers. Endomyseal fibrosis and fat ingrowth is evident (A, ×200, H&E). Macrophage infiltratioin of the endomysium, degenerating myofibers (B, ×200, H&E) and perivascular areas, but there are no vasculitis (C, ×200, H&E). Macrophage infiltration in myofibers is shown in H&E stain (D-1, ×400). Macrophages are positive for CD68 immunohistochemistry (D-2). There are a few CD3 positive T lymphocytes (D-3) but no B lymphocytes in CD20 stain (D-4).
jrd-19-285-g002

References

1. Gherardi RK, Coquet M, Chérin P, Authier FJ, Laforêt P, Bélec L, et al. Macrophagic myofasciitis: an emerging entity. Groupe dEtudes et Recherche sur les Maladies Musculaires Acquises et Dysimmunitaires (GERMMAD) de l'Association Française contre les Myopathies (AFM). Lancet. 1998. 352:347–352.
2. Authier FJ, Cherin P, Creange A, Bonnotte B, Ferrer X, Abdelmoumni A, et al. Central nervous system disease in patients with macrophagic myofasciitis. Brain. 2001. 124:974–983.
3. Cherin P, Gherardi RK. Macrophagic myofasciitis. Curr Rheumatol Rep. 2000. 2:196–200.
4. Gherardi RK, Coquet M, Cherin P, Belec L, Moretto P, Dreyfus PA, et al. Macrophagic myofasciitis lesions assess long-term persistence of vaccine-derived aluminium hydroxide in muscle. Brain. 2001. 124:1821–1831.
5. Ryan AM, Bermingham N, Harrington HJ, Keohane C. Atypical presentation of macrophagic myofasciitis 10 years post vaccination. Neuromuscul Disord. 2006. 16:867–869.
6. Park JH, Na KS, Park JH, Park YW, Paik SS, Yoo DH. Macrophagic myofasciitis unrelated to vaccination. Scand J Rheumatol. 2005. 34:65–67.
7. Rimaniol AC, Gras G, Verdier F, Capel F, Grigoriev VB, Porcheray F, et al. Aluminum hydroxide adjuvant induces macrophage differentiation towards a specialized antigen-presenting cell type. Vaccine. 2004. 22:3127–3135.
8. Guis S, Pellissier JF, Nicoli F, Reviron D, Mattei JP, Gherardi RK, et al. HLA-DRB1*01 and macrophagic myofasciitis. Arthritis Rheum. 2002. 46:2535–2537.
9. Guis S, Mattéi JP, Lioté F. Drug-induced and toxic myopathies. Best Pract Res Clin Rheumatol. 2003. 17:877–907.
10. 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.
11. Chérin P, Menard D, Mouton P, Viallard JF, Le Hello C, Authier FJ, et al. Macrophagic myofasciitis associated with inclusion body myositis: a report of three cases. Neuromuscul Disord. 2001. 11:452–457.
12. Schonberger LB, Bregman DJ, Sullivan-Bolyai JZ, Keenlyside RA, Ziegler DW, Retailliau HF, et al. Guillain-Barre syndrome following vaccination in the National Influenza Immunization Program, United States, 1976-1977. Am J Epidemiol. 1979. 110:105–123.
13. Geier DA, Geier MR. A case-control study of serious autoimmune adverse events following hepatitis B immunization. Autoimmunity. 2005. 38:295–301.
14. Verstraeten T, Descamps D, David MP, Zahaf T, Hardt K, Izurieta P, et al. Analysis of adverse events of potential autoimmune aetiology in a large integrated safety database of AS04 adjuvanted vaccines. Vaccine. 2008. 26:6630–6638.
15. Orbach H, Agmon-Levin N, Zandman-Goddard G. Vaccines and autoimmune diseases of the adult. Discov Med. 2010. 9:90–97.
TOOLS
Similar articles