Secondary Pulmonary Alveolar Proteinosis Associated with Hematological Malignancy: Three Cases Report and Literature Review_Chinese Journal of Respiratory and Critical Care Medicine

Authors：

ChenLulu , TianYaqiong ,  ZhangYingwei , QiuYuying , ZhaoQi , ZhuangYi , CaiHourong

Department of Respiratory Medicine, The Affiliated Drum Tower Hospital of Nanjing University, Nanjing, Jiangsu, 210008, China;

Corresponding author：

ZhangYingwei, Email: yingwei16@aliyun.com

Keywords：

Pulmonary alveolar proteinosis; Hematological malignancy; Diagnosis

DOI：

10.7507/1671-6205.2015141

Video：

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Abstract Full text Figures/Tables Video References Cited by

Objective To highlight the characteristics of secondary pulmonary alveolar proteinosis (PAP) associated with malignant hematological diseases. Methods The clinical data of three patients with secondary PAP were analyzed and the related literature was reviewed. Results Three patients were diagnosed with secondary PAP by exclusion of primary or autoimmune PAP and denied the history of inhalation of occupational dusts. Two patients with secondary PAP were associated with chronic myelocytic leukemia, and the third one was associated with myelodysplastic syndrome. The performance on HRCT of the PAP associated with hematological malignancy was different from the primary PAP. Three patients were pathologically diagonised by brochoalveolar lavage fluid. One patient was successfully treated with inhalation of granulocyte-macrophage colony-stimulating factor (GM-CSF). Conclusions Secondary PAP associated with hematological malignancy is very rare. The untypical HRCT is the main cause of misdiagnosis. Some patients may benefit from GM-CSF theatment.

Citation： ChenLulu, TianYaqiong, ZhangYingwei, QiuYuying, ZhaoQi, ZhuangYi, CaiHourong. Secondary Pulmonary Alveolar Proteinosis Associated with Hematological Malignancy: Three Cases Report and Literature Review. Chinese Journal of Respiratory and Critical Care Medicine, 2015, 14(6): 588-591. doi: 10.7507/1671-6205.2015141 Copy

1.	Rosen SH, Castleman B, Liebow AA. Pulmonary alveolar proteinosis. N Engl J Med,1958,258:1123-1142.
2.	蔡后荣,张英为.肺泡蛋白沉积症.见:蔡后荣,李惠萍,主编.实用间质性肺疾病.北京:人民卫生出版社,2010,193-203.
3.	Goldstein LS, Kavuru MS, Curtis-McCarthy P, et al. Pulmonary alveolar proteinosis-Clinical features and outcomes. Chest,1998,114:1357-1362.
4.	Thomassen MJ, Yi TL, Raychaudhuri B, et al. Pulmonary alveolar proteinosis is a disease of decreased availability of GM-CSF rather than an intrinsic cellular defect. Clinical Immunology,2000,95:85-92.
5.	Cummings KJ, Nakano M, Omae K, et al. Indium lung disease. Chest,2012,141:1512-1521.
6.	Birsak CA, Rossem RN, Nijhuis-Heddes JM, et al. Pulmonary alveolar proteinosis:a complication in patients with hematologic malignancy. Neth J Med,2000,56:193-197.
7.	Ishii H, Tazawa R, Kaneko C, et al. Clinical features of secondary pulmonary alveolar proteinosis:pre-mortem cases in Japan. Eur Respir J,2011,37:465-468.
8.	Dirksen U, Hattenhorst U, Schneider P, et al. Defective expression of granulocyte-macrophage colony-stimulating factor/interleukin-3/interleukin-5 receptor common beta chain in children with acute myeloid leukemia associated with respiratory failure. Blood,1998,92:1097-1103.
9.	Nishida A, Miyamoto A, Yamamaoto H, et al. Possible association of trisomy 8 with secondary pulmonary alveolar proteinosis in myelodysplastic syndrome. Am J Respir Criti Care Med,2011,184:279-280.
10.	蔡后荣,张英为,侯杰.碎石路征在肺弥漫性疾病影像诊断中的意义.国际呼吸杂志,2006,26:522-524.
11.	Ishii H, Trapnell BC, Tazawa R, et al. Comparative study of high-resolution CT findings between autoimmune and secondary pulmonary alveolar proteinosis. Chest,2009,136:1348-1355.
12.	Pamuk GE, Turgut B, Vural O, et al. Pulmonary alveolar proteinosis in a patient with acute lymphoid leukemia regression after G-CSF therapy. Leuk Lymphoma,2003,44:871-874.
13.	Ishii H, Seymour JF, Tazawa R, et al. Secondary pulmonary alveolar proteinosis complicating myelodysplastic syndrome results in worsening of prognosis:a retrospective cohort study in Japan:a retrospective cohort study in Japan. BMC Pulm Med,2014,14:37.
14.	Chung JH, Pipavath SJ, Myerson DH, et al. Secondary pulmonary alveolar proteinosis a confusing and potentially serious complication of hematologic malignancy. J Thorac Imaging,2009,24:115-118.
15.	Rodriguez-Luaces M, Lafuente A, Martin MP, et al. Haematopoietic transplantation in pulmonary alveolar proteinosis associated with chronic myelogenous leukaemia. Bone Marrow Transplant,1997,20:507-510.
16.	Ohmachi K, Ogiya D, Morita F, et al. Secondary pulmonary alveolar proteinosis in a patient with chronic myeloid leukemia in the accelerated phase. Tokai J Exp Clin Med,2008,33:146-149.
17.	Ito K, Iwabe K, Okai T, et al. Rapidly progressive pulmonary alveolar proteinosis in a patient with chronic myelogenous leukemia. Intern Med,1994,33:710-713.
18.	Tsushima K, Koyama S, Saitou H, et al. Pulmonary alveolar proteinosis in a patient with chronic myelogenous leukemia. Respiration,1999,66:173-175.

1. Rosen SH, Castleman B, Liebow AA. Pulmonary alveolar proteinosis. N Engl J Med,1958,258:1123-1142.
2. 蔡后荣,张英为.肺泡蛋白沉积症.见:蔡后荣,李惠萍,主编.实用间质性肺疾病.北京:人民卫生出版社,2010,193-203.
3. Goldstein LS, Kavuru MS, Curtis-McCarthy P, et al. Pulmonary alveolar proteinosis-Clinical features and outcomes. Chest,1998,114:1357-1362.
4. Thomassen MJ, Yi TL, Raychaudhuri B, et al. Pulmonary alveolar proteinosis is a disease of decreased availability of GM-CSF rather than an intrinsic cellular defect. Clinical Immunology,2000,95:85-92.
5. Cummings KJ, Nakano M, Omae K, et al. Indium lung disease. Chest,2012,141:1512-1521.
6. Birsak CA, Rossem RN, Nijhuis-Heddes JM, et al. Pulmonary alveolar proteinosis:a complication in patients with hematologic malignancy. Neth J Med,2000,56:193-197.
7. Ishii H, Tazawa R, Kaneko C, et al. Clinical features of secondary pulmonary alveolar proteinosis:pre-mortem cases in Japan. Eur Respir J,2011,37:465-468.
8. Dirksen U, Hattenhorst U, Schneider P, et al. Defective expression of granulocyte-macrophage colony-stimulating factor/interleukin-3/interleukin-5 receptor common beta chain in children with acute myeloid leukemia associated with respiratory failure. Blood,1998,92:1097-1103.
9. Nishida A, Miyamoto A, Yamamaoto H, et al. Possible association of trisomy 8 with secondary pulmonary alveolar proteinosis in myelodysplastic syndrome. Am J Respir Criti Care Med,2011,184:279-280.
10. 蔡后荣,张英为,侯杰.碎石路征在肺弥漫性疾病影像诊断中的意义.国际呼吸杂志,2006,26:522-524.
11. Ishii H, Trapnell BC, Tazawa R, et al. Comparative study of high-resolution CT findings between autoimmune and secondary pulmonary alveolar proteinosis. Chest,2009,136:1348-1355.
12. Pamuk GE, Turgut B, Vural O, et al. Pulmonary alveolar proteinosis in a patient with acute lymphoid leukemia regression after G-CSF therapy. Leuk Lymphoma,2003,44:871-874.
13. Ishii H, Seymour JF, Tazawa R, et al. Secondary pulmonary alveolar proteinosis complicating myelodysplastic syndrome results in worsening of prognosis:a retrospective cohort study in Japan:a retrospective cohort study in Japan. BMC Pulm Med,2014,14:37.
14. Chung JH, Pipavath SJ, Myerson DH, et al. Secondary pulmonary alveolar proteinosis a confusing and potentially serious complication of hematologic malignancy. J Thorac Imaging,2009,24:115-118.
15. Rodriguez-Luaces M, Lafuente A, Martin MP, et al. Haematopoietic transplantation in pulmonary alveolar proteinosis associated with chronic myelogenous leukaemia. Bone Marrow Transplant,1997,20:507-510.
16. Ohmachi K, Ogiya D, Morita F, et al. Secondary pulmonary alveolar proteinosis in a patient with chronic myeloid leukemia in the accelerated phase. Tokai J Exp Clin Med,2008,33:146-149.
17. Ito K, Iwabe K, Okai T, et al. Rapidly progressive pulmonary alveolar proteinosis in a patient with chronic myelogenous leukemia. Intern Med,1994,33:710-713.
18. Tsushima K, Koyama S, Saitou H, et al. Pulmonary alveolar proteinosis in a patient with chronic myelogenous leukemia. Respiration,1999,66:173-175.

Chinese Journal of Respiratory and Critical Care Medicine

Secondary Pulmonary Alveolar Proteinosis Associated with Hematological Malignancy: Three Cases Report and Literature Review

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