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Isolated Pauciimmune Pulmonary Capillaritis Successfully Treated With RituximabCapillaritis Successfully Treated With Rituximab

Gwen Thompson, MD; Ulrich Specks, MD; Rodrigo Cartin-Ceba, MD
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From the Department of Medicine (Drs Thompson, Specks, and Cartin-Ceba) and Division of Pulmonary and Critical Care Medicine (Drs Specks and Cartin-Ceba), Mayo Clinic, Rochester, MN.

CORRESPONDENCE TO: Rodrigo Cartin-Ceba, MD, Division of Pulmonary and Critical Care Medicine, Mayo Clinic, 200 First St SW, Rochester, MN 55905; e-mail: cartinceba.rodrigo@mayo.edu


This study was presented in abstract form at the American Thoracic Society Annual Meeting, San Diego, CA, May 16-21, 2014.

Reproduction of this article is prohibited without written permission from the American College of Chest Physicians. See online for more details.


Chest. 2015;147(4):e134-e136. doi:10.1378/chest.14-1884
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Diffuse alveolar hemorrhage (DAH) is a syndrome caused by different mechanisms, including capillary stress failure, diffuse alveolar damage, and capillaritis. Capillaritis is the most common cause and is often associated with systemic autoimmune disorders, most commonly antineutrophilic cytoplasmic antibody-associated vasculitis. The occurrence of DAH with underlying pulmonary capillaritis but without clinical or serologic findings of an associated underlying systemic disorder is known as isolated pauciimmune pulmonary capillaritis (IPPC), and only eight cases have been described in the literature. The mainstay of treatment of this rare condition has been cyclophosphamide and glucocorticoids. When cases are unresponsive to cyclophosphamide, there is no known alternative treatment. Herein, we describe a case of IPPC that failed cyclophosphamide treatment with recurrent DAH. Rituximab therapy was then initiated with no further evidence of recurrence. This case report suggests that rituximab could be considered an alternative therapy to induce remission in patients with IPPC.

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