J Rheum Dis.  2012 Oct;19(5):285-289. 10.4078/jrd.2012.19.5.285.

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

• Han M
• Park J
• Ryu H
• Choi IA
• Park W
• Lee EB
• Song YW
• Jung H
• Lee EY

• KMID: 1438538
• DOI: http://doi.org/10.4078/jrd.2012.19.5.285

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.

Keyword

Macrophagic myofasciitis; Vaccine; Steroid

MeSH Terms

Acute-Phase Proteins
Adult
Aluminum Hydroxide
Biopsy
Fasciitis
Female
Fever
Humans
Hydroxides
L-Lactate Dehydrogenase
Leukocytosis
Macrophages
Muscle Weakness
Muscles
Myositis
Physical Examination
Rare Diseases
Reference Values
Thigh
Vaccination
Vaccines
Acute-Phase Proteins
Aluminum Hydroxide
Fasciitis
Hydroxides
L-Lactate Dehydrogenase
Myositis
Vaccines

Figure

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  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.

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  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).

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