Abstract
Chronic organizing pneumonia (COP) has often been reported as a pulmonary manifestation of collagen vascular diseases, mainly rheumatoid arthritis, but the association of COP and dermatomyositis (DM) has rarely been documented. We report a 55 year-old woman with well-documented DM and a COP. She was refractory to steroids and two other immunosuppressive agents therapy (cyclophosphamide and azathioprine). Therefore, rituximab (2 x 1 g infusions) was used for treatment. During the following weeks her strength gradually increased while creatine kinase (CK), C reactive protein and erythrocyte sedimentation rate normalized. After 6 months, she had a relapse with increased muscle enzymes, fever and moderate muscle weakness. After a second course of rituximab (2 x 1 g infusions), the patient demonstrated a remarkable clinical response as indicated by an increase in muscle strength and moderate decline in creatine kinase levels. Lung abnormalities resolved significantly on high resolution chest CT scan. Thus, B-cell depletion therapy with rituximab used alone or in combination with other immunosuppressants may be a viable option in patients with polymyositis-dermatomyositis and pneumonia refractory to current therapies.
Highlights
Chronic organizing pneumonia (COP) has often been reported as a pulmonary manifestation of collagen vascular diseases, mainly rheumatoid arthritis, but the association of COP and dermatomyositis (DM) has rarely been documented
El compromiso pulmonar y las da, descrito por Epler en 1985, que se caracteriza complicaciones respiratorias son frecuentes en la por la presencia de focos de tejido conectivo laxo evolución de la enfermedad, entre las cuales se en la vía aérea distal asociado a inflamación incluyen la debilidad de los músculos respirato- crónica alveolar[6,7]
La radiografía de tórax en el momento del pecialmente en los lóbulos inferiores (Figura diagnóstico muestra opacidades intersticiales y de relleno 1) y el estudio funcional respiratorio mostraba alveolar bilaterales de predominio en ambos lóbulos una limitación espirométrica restrictiva y cainferiores
Summary
Chronic organizing pneumonia (COP) has often been reported as a pulmonary manifestation of collagen vascular diseases, mainly rheumatoid arthritis, but the association of COP and dermatomyositis (DM) has rarely been documented. PM/DM, siendo los principales patrones histológi- clínico de una paciente con dermatomiositis que cos la neumonía intersticial inespecífica (NSIP), consultó por síntomas respiratorios y compromiso neumonía intersticial usual (UIP), neumonía en pulmonar intersticial alrededor de un año antes organización (COP) y daño alveolar difuso (DAD).
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