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- Title
Rituximab for auto-immune alveolar proteinosis, a real life cohort study.
- Authors
Soyez, Berenice; Borie, Raphael; Menard, Cedric; Cadranel, Jacques; Chavez, Leonidas; Cottin, Vincent; Gomez, Emmanuel; Marchand-Adam, Sylvain; Leroy, Sylvie; Naccache, Jean-Marc; Nunes, Hilario; Reynaud-Gaubert, Martine; Savale, Laurent; Tazi, Abdellatif; Wemeau-Stervinou, Lidwine; Debray, Marie-Pierre; Crestani, Bruno
- Abstract
<bold>Background: </bold>Whole lung lavage is the current standard therapy for pulmonary alveolar proteinosis (PAP) that is characterized by the alveolar accumulation of surfactant. Rituximab showed promising results in auto-immune PAP (aPAP) related to anti-GM-CSF antibody.<bold>Methods: </bold>We aimed to assess efficacy of rituximab in aPAP in real life and all patients with aPAP in France that received rituximab were retrospectively analyzed.<bold>Results: </bold>Thirteen patients were included. No patients showed improvement 6 months after treatment, but, 4 patients (30%) presented a significant decrease of alveolar-arterial difference in oxygen after 1 year. One patient received lung transplantation and one patient was lost of follow-up within one year. Although a spontaneous improvement cannot be excluded in these 4 patients, improvement was more frequent in patients naïve to prior specific therapy and with higher level of anti-GM-CSF antibodies evaluated by ELISA. No serious adverse event was evidenced.<bold>Conclusions: </bold>These data do not support rituximab as a second line therapy for patients with refractory aPAP.
- Subjects
PULMONARY alveolar proteinosis; RITUXIMAB; TREATMENT effectiveness; LUNG transplantation; ENZYME-linked immunosorbent assay; THERAPEUTICS
- Publication
Respiratory Research, 2018, Vol 19, Issue 1, p74
- ISSN
1465-9921
- Publication type
journal article
- DOI
10.1186/s12931-018-0780-5