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Inhaled GM-CSF for Pulmonary Alveolar Proteinosis.
N Engl J Med
September 2019
From Niigata University Medical and Dental Hospital, Niigata (R.T., T.U., K.A., T.S., A.H., Takahiro Tanaka, T. Takada, N.K., K.N.), the Department of Respirology, Graduate School of Medicine, Chiba University, Chiba (M.A., K. Tatsumi), Kurashiki Municipal Hospital, Kurashiki (R.E., S. Kondoh), the Department of Clinical Medicine, Institute of Tropical Medicine, Nagasaki University, Nagasaki (K.M., Takeshi Tanaka), the Division of Respiratory Medicine and Allergology, Department of Medicine, Aichi Medical University School of Medicine, Aichi (E.Y., A.T.), the Department of Respiratory Medicine, Kyorin University School of Medicine (M.O., H.I.), and the Center Hospital of the National Center for Global Health and Medicine (S.I., H.S., A.M.), Tokyo, Kobe City Medical Center General Hospital, Kobe (A.N., K. Tomii), the Department of Respiratory Medicine, Faculty of Medicine and Graduate School of Medicine, Hokkaido University, Sapporo (M.S., S. Konno), the Department of Respiratory Medicine and Department of Occupational Health, Tohoku University Graduate School of Medicine, Sendai (S.O., N.T.), the Departments of Advanced Medicine for Respiratory Failure (T. Handa) and Respiratory Medicine (T. Handa, T. Hirai), Graduate School of Medicine, Kyoto University, Kyoto, and the National Hospital Organization Kinki-Chuo Chest Medical Center, Osaka (Y.I., T.A.) - all in Japan.
Background: Pulmonary alveolar proteinosis is a disease characterized by abnormal accumulation of surfactant in the alveoli. Most cases are autoimmune and are associated with an autoantibody against granulocyte-macrophage colony-stimulating factor (GM-CSF) that prevents clearing of pulmonary surfactant by alveolar macrophages. An open-label, phase 2 study showed some therapeutic efficacy of inhaled recombinant human GM-CSF in patients with severe pulmonary alveolar proteinosis; however, the efficacy in patients with mild-to-moderate disease remains unclear.
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