Rituximab for auto-immune alveolar proteinosis, a real life cohort study
Abstract Background 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. Methods We aimed to assess ef...
| Main Authors: | , , , , , , , , , , , , , , , , |
|---|---|
| Format: | Article |
| Language: | English |
| Published: |
BMC
2018-04-01
|
| Series: | Respiratory Research |
| Subjects: | |
| Online Access: | http://link.springer.com/article/10.1186/s12931-018-0780-5 |
| _version_ | 1828772560545775616 |
|---|---|
| author | Berenice Soyez Raphael Borie Cedric Menard Jacques Cadranel Leonidas Chavez Vincent Cottin Emmanuel Gomez Sylvain Marchand-Adam Sylvie Leroy Jean-Marc Naccache Hilario Nunes Martine Reynaud-Gaubert Laurent Savale Abdellatif Tazi Lidwine Wemeau-Stervinou Marie-Pierre Debray Bruno Crestani |
| author_facet | Berenice Soyez Raphael Borie Cedric Menard Jacques Cadranel Leonidas Chavez Vincent Cottin Emmanuel Gomez Sylvain Marchand-Adam Sylvie Leroy Jean-Marc Naccache Hilario Nunes Martine Reynaud-Gaubert Laurent Savale Abdellatif Tazi Lidwine Wemeau-Stervinou Marie-Pierre Debray Bruno Crestani |
| author_sort | Berenice Soyez |
| collection | DOAJ |
| description | Abstract Background 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. Methods 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. Results 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. Conclusions These data do not support rituximab as a second line therapy for patients with refractory aPAP. |
| first_indexed | 2024-12-11T14:46:13Z |
| format | Article |
| id | doaj.art-6f1f9ae9d0954af9b22b078bdbe8d1db |
| institution | Directory Open Access Journal |
| issn | 1465-993X |
| language | English |
| last_indexed | 2024-12-11T14:46:13Z |
| publishDate | 2018-04-01 |
| publisher | BMC |
| record_format | Article |
| series | Respiratory Research |
| spelling | doaj.art-6f1f9ae9d0954af9b22b078bdbe8d1db2022-12-22T01:01:40ZengBMCRespiratory Research1465-993X2018-04-011911710.1186/s12931-018-0780-5Rituximab for auto-immune alveolar proteinosis, a real life cohort studyBerenice Soyez0Raphael Borie1Cedric Menard2Jacques Cadranel3Leonidas Chavez4Vincent Cottin5Emmanuel Gomez6Sylvain Marchand-Adam7Sylvie Leroy8Jean-Marc Naccache9Hilario Nunes10Martine Reynaud-Gaubert11Laurent Savale12Abdellatif Tazi13Lidwine Wemeau-Stervinou14Marie-Pierre Debray15Bruno Crestani16Service de Pneumologie A, DHU FIRE, centre de référence constitutif des maladies pulmonaires rares, Hôpital Bichat, APHPService de Pneumologie A, DHU FIRE, centre de référence constitutif des maladies pulmonaires rares, Hôpital Bichat, APHPService d’Immunologie, Thérapie Cellulaire et Hématopoïèse, CHU PontchaillouOrphaLungService de Pneumologie, Centre de compétences des maladies pulmonaires rares, CHU Grenoble-AlpesOrphaLungOrphaLungOrphaLungOrphaLungOrphaLungOrphaLungOrphaLungService de Pneumologie, Centre de référence de l’hypertension pulmonaire, Hôpital Bicêtre, APHPService de Pneumologie, Hôpital Saint-Louis, APHPOrphaLungService de Radiologie, Hôpital Bichat, APHPService de Pneumologie A, DHU FIRE, centre de référence constitutif des maladies pulmonaires rares, Hôpital Bichat, APHPAbstract Background 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. Methods 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. Results 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. Conclusions These data do not support rituximab as a second line therapy for patients with refractory aPAP.http://link.springer.com/article/10.1186/s12931-018-0780-5Whole lung lavageGM-CSFSurfactantInterstitial lung diseaseTherapy |
| spellingShingle | Berenice Soyez Raphael Borie Cedric Menard Jacques Cadranel Leonidas Chavez Vincent Cottin Emmanuel Gomez Sylvain Marchand-Adam Sylvie Leroy Jean-Marc Naccache Hilario Nunes Martine Reynaud-Gaubert Laurent Savale Abdellatif Tazi Lidwine Wemeau-Stervinou Marie-Pierre Debray Bruno Crestani Rituximab for auto-immune alveolar proteinosis, a real life cohort study Respiratory Research Whole lung lavage GM-CSF Surfactant Interstitial lung disease Therapy |
| title | Rituximab for auto-immune alveolar proteinosis, a real life cohort study |
| title_full | Rituximab for auto-immune alveolar proteinosis, a real life cohort study |
| title_fullStr | Rituximab for auto-immune alveolar proteinosis, a real life cohort study |
| title_full_unstemmed | Rituximab for auto-immune alveolar proteinosis, a real life cohort study |
| title_short | Rituximab for auto-immune alveolar proteinosis, a real life cohort study |
| title_sort | rituximab for auto immune alveolar proteinosis a real life cohort study |
| topic | Whole lung lavage GM-CSF Surfactant Interstitial lung disease Therapy |
| url | http://link.springer.com/article/10.1186/s12931-018-0780-5 |
| work_keys_str_mv | AT berenicesoyez rituximabforautoimmunealveolarproteinosisareallifecohortstudy AT raphaelborie rituximabforautoimmunealveolarproteinosisareallifecohortstudy AT cedricmenard rituximabforautoimmunealveolarproteinosisareallifecohortstudy AT jacquescadranel rituximabforautoimmunealveolarproteinosisareallifecohortstudy AT leonidaschavez rituximabforautoimmunealveolarproteinosisareallifecohortstudy AT vincentcottin rituximabforautoimmunealveolarproteinosisareallifecohortstudy AT emmanuelgomez rituximabforautoimmunealveolarproteinosisareallifecohortstudy AT sylvainmarchandadam rituximabforautoimmunealveolarproteinosisareallifecohortstudy AT sylvieleroy rituximabforautoimmunealveolarproteinosisareallifecohortstudy AT jeanmarcnaccache rituximabforautoimmunealveolarproteinosisareallifecohortstudy AT hilarionunes rituximabforautoimmunealveolarproteinosisareallifecohortstudy AT martinereynaudgaubert rituximabforautoimmunealveolarproteinosisareallifecohortstudy AT laurentsavale rituximabforautoimmunealveolarproteinosisareallifecohortstudy AT abdellatiftazi rituximabforautoimmunealveolarproteinosisareallifecohortstudy AT lidwinewemeaustervinou rituximabforautoimmunealveolarproteinosisareallifecohortstudy AT mariepierredebray rituximabforautoimmunealveolarproteinosisareallifecohortstudy AT brunocrestani rituximabforautoimmunealveolarproteinosisareallifecohortstudy |