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To the Editor: The recent report by Kokotis, et al1, who describe bilateral ocular myositis occurring in a patient several years after dermatomyositis (DM), prompted us to report the following case. A 71-year-old woman presented in December 1999 with a 3-month history of proptosis, initially unilateral but rapidly involving both eyes. Ten years earlier she developed muscular fatigue and weakness of upper and lower limbs, and an inflammatory myopathy was diagnosed by muscle biopsy. She was started on steroids, which were withdrawn after several months for severe osteoporosis; a subsequent course of cyclophosphamide was spontaneously discontinued because of well being. In June 1999, she was found to have atrial fibrillation and systemic arterial hypertension and was given digoxin, diuretics, and angiotensin-converting enzyme inhibitors. On admission, physical examination was unremarkable, except for bilateral proptosis and exophthalmus; blood pressure was normal, atrial fibrillation was present with heart rate of 80/min. Laboratory evaluation showed antinuclear antibodies (ANA, 1:640, homogeneous pattern), but no other autoantibodies (anti-DNA, antiextractable nuclear antigen, antineutrophil cytoplasmic antibodies, anticardiolipin, and rheumatoid factor). The other laboratory data were normal, including creatine phosphokinase, C-reactive protein, complement, and thyroid function tests. Orbital magnetic resonance imaging (MRI) was consistent with inflammatory myopathy of extrinsic ocular muscles. Electromyography (EMG) of arm and leg muscles showed mild involvement confined to deltoid. She refused azathioprine and began methotrexate (MTX), 7.5 mg once a week, with a slow but significant improvement. Orbital MRI was unchanged in June 2000, but by December 2000 did not show any myopathy, even though a mild exophthalmus persisted. MTX was progressively reduced and discontinued. When last seen (March 2004), she was in good health. As far as we know, this is the second case of bilateral orbital myositis in patients with systemic inflammatory myopathy. Interestingly, our patient, like the patient described by Kokotis, et al1, developed orbital myositis several years after systemic muscle involvement, with no other symptoms than proptosis and exophthalmus, and with normal circulating muscle enzymes (Kokotis's patient reported erythema on the back and eyelid). Several cases of orbital myositis have been reported in association with autoimmune rheumatic diseases2-4, however, although our patient had antinuclear antibodies, since there were no symptoms/signs suggesting other rheumatic diseases, since mild signs of myopathy were indeed recorded on EMG, and because of the previous biopsy-based diagnosis of inflammatory polymyositis, we conclude that our patient had bilateral ocular myositis as a late complication of polymyositis. Another interesting feature that seemed to emerge in both patients was good response to low dose MTX. Our patient was started on MTX, having refused azathioprine and because steroids were contraindicated for severe osteoporosis. Kokotis's patient did not benefit from azathioprine or intravenous immunoglobulin, but did improve with MTX. Poor response to steroids has been recently reported in a child with idiopathic orbital myositis5. In accord with Kokotis, et al1, we conclude that ocular myositis is probably less rare than previously thought and suggest the usefulness of low dose MTX. BIANCA MARASINI, MD, Department of Medicine, Surgery and Dentistry, Rheumatology Unit, Istituto Clinico Humanitas, University of Milan; ROBERTA COSSUTTA, MD, Rheumatology Unit, Istituto Clinico Humanitas; MARCO MASSAROTTI, MD, Rheumatology Unit, Istituto Clinico Humanitas, Milan, Italy. Address reprint requests to Dr. B. Marasini, Istituto Clinico Humanitas, Via Manzoni 56, 20089 Rozzano, Milan, Italy REFERENCES 1. Kokotis P, Theodossiadis P, Kouros C, Sfikakis PP. Bilateral ocular myositis as a late complication of dermatomyositis. J Rheumatol 2005;32:379-81. 2. Billing K, Malhotra R, Selva D, Dodd T. Orbital myositis in Churg-Strauss syndrome. Acta Ophthalmol 2004;122:393-6. 3. Younes M, Bejia I, Moussa A, et al. Oculomotor muscle involvement revealing dermatomyositis in a patient with rheumatoid arthritis. Rev Med Interne 2004;25:829-32. 4. Tsironi E, Eftaxias B, Karabatsas CH, Ioachim E, Kalogeropoulos C, Psilas K. An unusually longstanding, strictly ocular, limited form of Wegener's granulomatosis. Acta Ophthalmol 2005; 83:123-5. 5. Hattori H, Ohnishi S, Nakagawa Y, et al. An infant with idiopathic orbital myositis poorly responsive to steroid therapy: a case report. Brain Dev 2005;27:160-2. |