minocycline and Bronchiolitis-Obliterans

Bronchiolitis obliterans is a small-airway obstructive lung disease for which immunologically mediated pathogenesis is supposed. Frequent association of bronchiolitis obliterans with paraneoplastic pemphigus is well known, but its association with other autoimmune bullous diseases has not been reported except for a case of anti-laminin-332-type mucous membrane pemphigoid in a patient with chronic graft-versus-host disease. We report a case of non-paraneoplastic autoimmune subepidermal bullous disease associated with fatal bronchiolitis obliterans in a patient without transplantation. Although the patient's serum contained immunoglobulin (Ig)A antibodies to the 180-kDa bullous pemphigoid antigen/type XVII collagen and IgG antibodies to laminin-332, diagnosis of either linear IgA bullous dermatosis or mucous membrane pemphigoid could not be made because of the failure to detect linear IgA deposition at the basement membrane zone by direct immunofluorescence and the lack of mucous membrane lesions. Physicians should be aware that autoimmune bullous diseases other than paraneoplastic pemphigus can also associate with this rare but potentially fatal lung disease.

Topics: Aged; Anti-Bacterial Agents; Autoantibodies; Autoantigens; Basement Membrane; Biopsy; Bronchiolitis Obliterans; Cell Adhesion Molecules; Collagen Type XVII; Fatal Outcome; Fluorescent Antibody Technique, Direct; Glucocorticoids; Humans; Immunoglobulin A; Immunoglobulin G; Kalinin; Male; Minocycline; Niacinamide; Non-Fibrillar Collagens; Paraneoplastic Syndromes; Pemphigoid, Bullous; Prednisolone; Respiratory Insufficiency; Vitamin B Complex

We discribe a rare case of rheumatoid arthritis (RA) complicated with bronchiolitis obliterans that was successfully treated with minocycline. Sixty four-year old woman with a four-years history of RA was admitted to the hospital because of dyspnea on exertion and polyarthritis. Pulmonary function test revealed marked decrease in V25 (0.10 l/s: 6.9%) and MMFR (12.6%). High resolution CT of the lung showed scattered centri-lobular micronodules in both lung fields, mucoid impaction, and hyperinflation. These findings indicated the presence of bronchiolitis obliterans. After 3 months of the treatment with minocycline, the patient showed a significant improvement of both arthritis and pulmonary function. Chest CT findings also improved after 1 year. The present case suggests that minocycline is effective for the treatment of bronchiolitis obliterans seen in patients with RA.

Topics: Anti-Bacterial Agents; Arthritis, Rheumatoid; Bronchiolitis Obliterans; Female; Humans; Middle Aged; Minocycline