{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T00:51:44Z","timestamp":1791161504328,"version":"4.1.0"},"reference-count":43,"publisher":"Wiley","issue":"10","license":[{"start":{"date-parts":[[2026,7,15]],"date-time":"2026-07-15T00:00:00Z","timestamp":1784073600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,7,15]],"date-time":"2026-07-15T00:00:00Z","timestamp":1784073600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/501100001659","name":"Deutsche Forschungsgemeinschaft","doi-asserted-by":"publisher","award":["DFG 970\/9\u20102"],"award-info":[{"award-number":["DFG 970\/9\u20102"]}],"id":[{"id":"10.13039\/501100001659","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["R01 HL085453"],"award-info":[{"award-number":["R01 HL085453"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Respirology"],"published-print":{"date-parts":[[2026,10]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background and Objective<\/jats:title>\n                    <jats:p>Pulmonary alveolar proteinosis (PAP) is a rare disease of inappropriate alveolar surfactant accumulation. The study objective was to compare Greece\u2013T\u00fcrkiye\u2013Cyprus cohorts, followed\u2010up for 20\u2009years.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Data were retrieved retrospectively by chart review.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      One hundred and thirty patients, Greece 39, T\u00fcrkiye 87, Cyprus 4, were included; 115 (89%) autoimmune (a)PAP, 13 (10%) secondary, 1 (1%) hereditary. Abnormal anti\u2010GM\u2010CSF antibody titre was available in 86\/115 patients, further analyzed for aPAP. Forty (46%) were male, with median (IQR) age at diagnosis of 40 (31\u201350) years. At median (IQR) duration of clinical follow\u2010up of 39 (18\u201378) months, three patients died. Non\u2010survivors had higher rates of cardiovascular disease [67% vs. 5%,\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&lt;\u20090.001], LTOT (67% vs. 13%,\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.001), pulmonary fibrosis [67% vs. 5%] and worse functional status at follow\u2010up [FVC% pred 49 (29) vs. 87 (74\u201397);\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.026, DLCO% pred 21 (46) vs. 72 (58\u201382);\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.016]. aPAP patients were stratified by therapy group (no WLL\u2010no i\u2010GM\u2010CSF, WLL alone, i\u2010GMCSF alone and both WLL and iGM\u2010CSF). WLL was more frequently performed in T\u00fcrkiye and i\u2010GMCSF alone in Greece (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.006). All therapies were effective. No difference was detected on outcome when sole iGM\u2010CSF was compared to WLL alone or in combination with iGM\u2010CSF [deceased\/alive 0%\/100% vs. 7%\/93%,\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.491]. Sole iGM\u2010CSF significantly ameliorated clinical and functional parameters of disease severity [DLCO% (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.013), SatO\n                      <jats:sub>2<\/jats:sub>\n                      (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.002), 6MWT (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.026)]. Across all therapy groups, pulmonary fibrosis eliminated beneficial response to treatment.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>This first real\u2010life comparison of aPAP cohorts originating from three countries showed the non\u2010inferiority of sole iGM\u2010CSF treatment. Fibrosis negatively affects response to treatment and survival.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/resp.70288","type":"journal-article","created":{"date-parts":[[2026,7,16]],"date-time":"2026-07-16T00:09:09Z","timestamp":1784160549000},"page":"1014-1028","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Pulmonary Alveolar Proteinosis in Greece\u2013T\u00fcrkiye\u2013Cyprus: Answers in a Real\u2010Life Comparison"],"prefix":"10.1002","volume":"31","author":[{"given":"Spyros A.","family":"Papiris","sequence":"first","affiliation":[{"name":"Medical School, National and Kapodistrian University of Athens  Athens Greece"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Efsun Gonca U\u011fur","family":"Chousein","sequence":"additional","affiliation":[{"name":"Yedikule Chest Diseases and Thoracic Surgery, Education and Research Hospital University of Health Sciences  \u0130stanbul T\u00fcrkiye"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maria","family":"Kallieri","sequence":"additional","affiliation":[{"name":"2nd Pulmonary Medicine Department General University Hospital \u201cAttikon\u201d, Medical School, National and Kapodistrian University of Athens  Athens Greece"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1870-5669","authenticated-orcid":false,"given":"Nesrin","family":"Mogulkoc","sequence":"additional","affiliation":[{"name":"Department of Pulmonary Medicine Ege University Medical School, Ege University Hospital  Izmir T\u00fcrkiye"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andriana I.","family":"Papaioannou","sequence":"additional","affiliation":[{"name":"1st Respiratory Medicine Department \u201cSotiria\u201d Hospital, National and Kapodistrian University of Athens  Athens Greece"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frangiskos","family":"Frangopoulos","sequence":"additional","affiliation":[{"name":"Pulmonary Medicine Department Larnaca General Hospital  Larnaca Cyprus"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tonia","family":"Adamide","sequence":"additional","affiliation":[{"name":"Respiratory Clinic, General Hospital of Nicosia  Nicosia Cyprus"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lykourgos","family":"Kolilekas","sequence":"additional","affiliation":[{"name":"7th Pulmonary Department Athens Chest Hospital \u201cSotiria\u201d  Athens Greece"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Burak","family":"G\u00f6k\u00e7e","sequence":"additional","affiliation":[{"name":"Department of Pulmonary Medicine Ege University Medical School, Ege University Hospital  Izmir T\u00fcrkiye"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nesrin","family":"Ocal","sequence":"additional","affiliation":[{"name":"Department of Pulmonology Gulhane Faculty of Medicine  Ankara T\u00fcrkiye"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Despina","family":"Papakosta","sequence":"additional","affiliation":[{"name":"Department of Pulmonary Medicine Aristotle University of Thessaloniki, \u201cG. 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