{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,21]],"date-time":"2026-07-21T17:35:36Z","timestamp":1784655336015,"version":"3.55.0"},"reference-count":21,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2022,10,25]],"date-time":"2022-10-25T00:00:00Z","timestamp":1666656000000},"content-version":"vor","delay-in-days":297,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100019018","name":"Chinese Academy of Medical Sciences Initiative for Innovative Medicine","doi-asserted-by":"publisher","award":["2017-12\u2009M-2-001"],"award-info":[{"award-number":["2017-12\u2009M-2-001"]}],"id":[{"id":"10.13039\/501100019018","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100012166","name":"National Key Research and Development Program of China","doi-asserted-by":"publisher","award":["2016YFC0901502"],"award-info":[{"award-number":["2016YFC0901502"]}],"id":[{"id":"10.13039\/501100012166","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["81570061"],"award-info":[{"award-number":["81570061"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["BioMed Research International"],"published-print":{"date-parts":[[2022,1]]},"abstract":"<jats:p><jats:italic>Background<\/jats:italic>. Pulmonary alveolar proteinosis (PAP) is a rare disorder which is characterized by the accumulation of excessive surfactant lipids and proteins in alveolar macrophages and alveoli. Oral statin therapy has been reported to be a novel therapy for PAP with hypercholesterolemia. We aimed to evaluate the safety and efficacy of oral statin therapy for PAP without hypercholesterolemia. <jats:italic>Methods<\/jats:italic>. In a prospective real\u2010world observational study, 47 PAP patients without hypercholesterolemia were screened. Oral statin was initiated as therapy for these PAP patients with 12 months of follow\u2010up. <jats:italic>Results<\/jats:italic>. Forty PAP patients completed the study. 26 (65%) of 40 PAP patients responded to statin therapy according to the study criteria. Partial pressure of arterial oxygen (PaO2) and percentage of diffusion capacity predicted (DLCO%) significantly increased while disease severity score (DSS) and radiographic abnormalities decreased after 12 months of statin therapy (all <jats:italic>p<\/jats:italic> &lt; 0.05). The factors associated with response were higher levels of granulocyte\u2010macrophage colony\u2010stimulating factor (GM\u2010CSF) antibody and baseline total cholesterol\/high\u2010density lipoprotein cholesterol (TC\/HDL) (<jats:italic>p<\/jats:italic> = 0.015 and <jats:italic>p<\/jats:italic> = 0.035, respectively). The area under the receiver operating characteristic curve (AUROC) of dose of atorvastatin for predicting the response to statin therapy for PAP was 0.859 (95% CI: 0.738\u20100.979, <jats:italic>p<\/jats:italic> &lt; 0.001). The cutoff dose of atorvastatin was 67.5\u2009mg daily with their corresponding specificity (64.3%) and sensitivity (96.2%). No severe side effects were observed during the study. <jats:italic>Conclusions<\/jats:italic>. In PAP patients without hypercholesterolemia, statin therapy resulted in improvements in arterial blood gas (ABG) measurement, pulmonary function, and radiographic assessment.<\/jats:p>","DOI":"10.1155\/2022\/1589660","type":"journal-article","created":{"date-parts":[[2022,10,25]],"date-time":"2022-10-25T21:20:05Z","timestamp":1666732805000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":18,"title":["Assessment of Statin Treatment for Pulmonary Alveolar Proteinosis without Hypercholesterolemia: A 12\u2010Month Prospective, Longitudinal, and Observational Study"],"prefix":"10.1155","volume":"2022","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2022-121X","authenticated-orcid":false,"given":"Shenyun","family":"Shi","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2768-3126","authenticated-orcid":false,"given":"Xianhua","family":"Gui","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9700-4504","authenticated-orcid":false,"given":"Jingjing","family":"Ding","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8178-4948","authenticated-orcid":false,"given":"Shangwen","family":"Yang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3462-8835","authenticated-orcid":false,"given":"Xiaoyan","family":"Xin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7662-531X","authenticated-orcid":false,"given":"Kaifeng","family":"Xu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1194-1394","authenticated-orcid":false,"given":"Yonglong","family":"Xiao","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2022,10,25]]},"reference":[{"key":"e_1_2_13_1_2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMra023226"},{"key":"e_1_2_13_2_2","doi-asserted-by":"publisher","DOI":"10.1038\/s41572-019-0066-3"},{"key":"e_1_2_13_3_2","doi-asserted-by":"publisher","DOI":"10.1183\/09031936.04.00102704"},{"key":"e_1_2_13_4_2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa1913590"},{"key":"e_1_2_13_5_2","doi-asserted-by":"publisher","DOI":"10.1038\/s41467-018-05491-z"},{"key":"e_1_2_13_6_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.jim.2013.11.011"},{"key":"e_1_2_13_7_2","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2003-05-1565"},{"key":"e_1_2_13_8_2","first-page":"497","article-title":"Serum markers in patients with idiopathic pulmonary alveolar proteinosis","volume":"37","author":"Li Y.","year":"2014","journal-title":"Zhonghua Jie He He Hu Xi Za Zhi= Zhonghua Jiehe He Huxi Zazhi= Chinese Journal of Tuberculosis and Respiratory Diseases"},{"key":"e_1_2_13_9_2","doi-asserted-by":"publisher","DOI":"10.1164\/rccm.200708-1271OC"},{"key":"e_1_2_13_10_2","doi-asserted-by":"publisher","DOI":"10.1136\/thorax.58.3.252"},{"key":"e_1_2_13_11_2","first-page":"209","article-title":"Granulocyte macrophage colony stimulating factor therapy for pulmonary alveolar proteinosis","volume":"61","author":"Shende R. 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