BAL findings in a patient with pulmonary alveolar proteinosis successfully treated with GM-CSF

Schoch, O. D.; Schanz, U.; Koller, M.; Nakata, K.; Seymour, J. F.; Russi, E. W.; Boehler, A.
March 2002
Thorax;Mar2002, Vol. 57 Issue 3, p277
Academic Journal
Background: ldiopathic pulmonary alveolar proteinosis (PAP) has recently been recognised as a disease of impaired alveolar macrophage function caused by neutralising antigranutocyte-macrophage colony-stimulating (anti-GM-CSF) autoantibodies. Subcutaneous recombinant human GM-CSF is a novel treatment for PAP, but its mechanism of action is unclear. Methods: Clinical functional, and bronchoalveolar lavage (BAL) findings were prospectively evaluated in a patient with PAP treated with daily subcutaneous GM-CSF 8 μg/kg for 12 weeks. Result: Treatment resulted in improvement in dyspnoea, lung function, and peak cycle ergometry performance. In serum and BAL fluid the titre of anti-GM-CSF autoantibodies was raised at baseline and markedly reduced on treatment. At baseline the BAL fluid cellular profile showed a decrease in the absolute number and the percentage of macrophages (50%) and an increase in lymphocytes (45%), predominantly CD4+. This cellular distribution remained unchanged after 6 and 12 weeks of treatment while macrophages became morphologically normal and functionally improved. Extracellular proteinaceous material completely disappeared. Conclusions: Clinically successful treatment of PAP with GM-CSF was associate profound reduction in GM-CSF neutralising autoantibodies, improvement in alveolar macrophage morphology and function, but persistent BAL lymphocytosis.


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