DOI QR코드

DOI QR Code

GM-CSF 흡입치료로 호전된 폐포단백증 1예

A Case of Pulmonary Alveolar Proteinosis Improved with Inhaled Granulocyte-Macrophage Colony-Stimulating Factor (GM-CSF)

  • 강보형 (울산대학교 의과대학 서울아산병원 내과) ;
  • 이호수 (울산대학교 의과대학 서울아산병원 내과) ;
  • 이유미 (울산대학교 의과대학 서울아산병원 내과) ;
  • 박소은 (울산대학교 의과대학 서울아산병원 내과) ;
  • 김우성 (울산대학교 의과대학 서울아산병원 호흡기내과) ;
  • 김동순 (울산대학교 의과대학 서울아산병원 호흡기내과) ;
  • 송진우 (울산대학교 의과대학 서울아산병원 호흡기내과)
  • Kang, Bo-Hyoung (Department of Internal Medicine, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Lee, Ho-Su (Department of Internal Medicine, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Lee, Yu-Mi (Department of Internal Medicine, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Park, So-Eun (Department of Internal Medicine, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Kim, Woo-Sung (Department of Pulmonary and Critical Care Medicine, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Kim, Dong-Soon (Department of Pulmonary and Critical Care Medicine, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Song, Jin-Woo (Department of Pulmonary and Critical Care Medicine, Asan Medical Center, University of Ulsan College of Medicine)
  • 발행 : 2012.03.01

초록

폐포단백증은 폐포에 표면활성제 기원의 인지질이 비정상적으로 다량 침착되는 드문 질병으로 자연 관해에서 호흡부전으로 인한 사망까지 다양한 임상경과를 보인다. 폐포단백증의 치료로는 현재까지 전폐 세척술이 표준치료로 받아 들여지고 있다. 하지만 전신마취 및 기관삽관 등이 필요한 침습적 시술이라는 위험성이 있어 최근에는 GM-CSF를 이용한 치료법들이 연구되고 있다. 본 증례는 내원 4년 전 폐포단백증으로 진단받은 환자로 호흡곤란 및 방사선학적 소견의 악화로 전폐 세척술을 4차례 시행 받았으나 잦은 재발로 GM-CSF 흡입치료를 시작한 환자로 흡입치료 후 증상의 호전 및 방사선학적 소견을 경험한 예이다.

Pulmonary alveolar proteinosis (PAP) is a rare lung disease characterized by the accumulation of lipoproteinaceous material within the alveoli. Several studies have recently found that autoantibodies against granulocyte-macrophage colony-stimulating factor (GM-CSF) play a major role in the pathogenesis of idiopathic PAP. Consequently, inhaled or systemic injection of GM-CSF has been suggested as a promising treatment for PAP. A 54-year-old male visited our hospital for progressive dyspnea. Four years earlier, he was diagnosed with PAP based on a surgical lung biopsy in another institution. Whole-lung lavage was performed four times before he visited our hospital. We administered high-dose inhaled GM-CSF therapy for 12 weeks followed by 12 weeks of low-dose therapy. After the GM-CSF treatment, the patient's symptoms, lung function, and radiological findings were improved significantly.

키워드

참고문헌

  1. Seymour JF, Presneill JJ. Pulmonary alveolar proteinosis: progress in the first 44 years. Am J Respir Crit Care Med 2002; 166:215-235. https://doi.org/10.1164/rccm.2109105
  2. Beccaria M, Luisetti M, Rodi G, et al. Long-term durable benefit after whole lung lavage in pulmonary alveolar proteinosis. Eur Respir J 2004;23:526-531. https://doi.org/10.1183/09031936.04.00102704
  3. Stanley E, Lieschke GJ, Grail D, et al. Granulocyte/macrophage colony-stimulating factor-deficient mice show no major perturbation of hematopoiesis but develop a characteristic pulmonary pathology. Proc Natl Acad Sci U S A 1994;91:5592-5596. https://doi.org/10.1073/pnas.91.12.5592
  4. Seymour JF, Presneill JJ, Schoch OD, et al. Therapeutic efficacy of granulocyte-macrophage colony-stimulating factor in patients with idiopathic acquired alveolar proteinosis. Am J Respir Crit Care Med 2001;163:524-531. https://doi.org/10.1164/ajrccm.163.2.2003146
  5. Song JW, Park SH, Kang KW. A case of idiopathic pulmonary alveolar proteinosis treated with granulocyte-macrophage colony stimulating factor (GM-CSF) after partial response to whole lung lavage. Tuberc Respir Dis 2009;67:569-573. https://doi.org/10.4046/trd.2009.67.6.569
  6. Tazawa R, Trapnell BC, Inoue Y, et al. Inhaled granulocyte/ macrophage-colony stimulating factor as therapy for pulmonary alveolar proteinosis. Am J Respir Crit Care Med 2010;181:1345-1354. https://doi.org/10.1164/rccm.200906-0978OC
  7. Chang B, Noh J, Ok CS, et al. A case of pulmonary alveolar proteinosis that improved with GM-CSF inhalation therapy. Korean J Med 2011;80:588-594.
  8. Kitamura T, Tanaka N, Watanabe J, et al. Idiopathic pulmonary alveolar proteinosis as an autoimmune disease with neutralizing antibody against granulocyte/macrophage colony-stimulating factor. J Exp Med 1999;190:875-880. https://doi.org/10.1084/jem.190.6.875
  9. Venkateshiah SB, Yan TD, Bonfield TL, et al. An open-label trial of granulocyte macrophage colony stimulating factor therapy for moderate symptomatic pulmonary alveolar proteinosis. Chest 2006;130:227-237. https://doi.org/10.1378/chest.130.1.227
  10. Wylam ME, Ten R, Prakash UB, Nadrous HF, Clawson ML, Anderson PM. Aerosol granulocyte-macrophage colony-stimulating factor for pulmonary alveolar proteinosis. Eur Respir J 2006;27: 585-593. https://doi.org/10.1183/09031936.06.00058305