{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,9]],"date-time":"2026-09-09T13:20:40Z","timestamp":1788960040475,"version":"build-2803163510"},"reference-count":39,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2020,10,23]],"date-time":"2020-10-23T00:00:00Z","timestamp":1603411200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Histopathology"],"published-print":{"date-parts":[[2021,2]]},"abstract":"<jats:sec>\n                    <jats:title>Aims<\/jats:title>\n                    <jats:p>Mucinous adenocarcinoma arising in congenital pulmonary airway malformation (CPAM) is a rare complication, with little being known about its natural course. The aims of this article are to describe a series of mucinous adenocarcinomas arising from CPAMs, and present their clinicopathological features, genetics, and clinical outcome.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods and results<\/jats:title>\n                    <jats:p>\n                      Thirty\u2010seven cases were collected within a 34\u2010year period, and the subtype of adenocarcinoma and CPAM, tumour location, stage, growth patterns, molecular data and follow\u2010up were recorded. The cohort comprised CPAM type 1 (\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u00a033) and CPAM type 2 (\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u00a04). Morphologically, 34 cases were mucinous adenocarcinomas (21\n                      <jats:italic>in\u00a0situ<\/jats:italic>\n                      ; 13 invasive), and three were mixed mucinous and non\u2010mucinous adenocarcinoma. Seventeen cases showed purely extracystic (intra\u2010alveolar) adenocarcinoma, 15 were mixed intracystic and extracystic, and five showed purely intracystic proliferation. Genetically, nine of 10 cases tested positive for\n                      <jats:italic>KRAS<\/jats:italic>\n                      mutations, four with exon 2 G12V mutation and five with exon 2 G12D mutation. Residual disease on completion lobectomy was observed in two cases, and three cases recurred 7, 15 and 32\u00a0years after the original diagnosis. Two patients died of metastatic invasive mucinous adenocarcinoma.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Most adenocarcinoma that arise in type 1 CPAMs, are purely mucinous, and are early\u2010stage disease. Intracystic proliferation is associated with lepidic growth, an absence of invasion, and indolent behaviour, whereas extracystic proliferation may be associated with more aggressive behaviour and advanced stage. Most cases are cured by lobectomy, and recurrence\/residual disease seems to be associated with limited surgery. Long\u2010term follow\u2010up is needed, as recurrence can occur decades later.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/his.14239","type":"journal-article","created":{"date-parts":[[2020,8,27]],"date-time":"2020-08-27T18:26:22Z","timestamp":1598552782000},"page":"434-444","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":46,"title":["Mucinous adenocarcinoma arising in congenital pulmonary airway malformation: clinicopathological analysis of 37 cases"],"prefix":"10.1111","volume":"78","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1005-7815","authenticated-orcid":false,"given":"Wei\u2010Chin","family":"Chang","sequence":"first","affiliation":[{"name":"Department of Histopathology Royal Brompton and Harefield NHS Foundation Trust London UK"},{"name":"Department of Pathology MacKay Memorial Hospital Taipei Taiwan"},{"name":"Department of Medicine MacKay Medical College, New Taipei City Taipei Taiwan"},{"name":"Institute of Clinical Medicine National Yang\u2010Ming University Taipei Taiwan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6134-980X","authenticated-orcid":false,"given":"Yu Zhi","family":"Zhang","sequence":"additional","affiliation":[{"name":"Department of Histopathology Royal Brompton and Harefield NHS Foundation Trust London UK"},{"name":"National Heart and Lung Institute Imperial College London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Janina L","family":"Wolf","sequence":"additional","affiliation":[{"name":"Department of Pathology Erasmus Medical Centre Sophia Children\u2019s Hospital Rotterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sergei M","family":"Hermelijn","sequence":"additional","affiliation":[{"name":"Department of Paediatric Surgery Erasmus Medical Centre Sophia Children\u2019s Hospital Rotterdam The 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Cancer Centre of L\u00e9on B\u00e9rard Lyon France"},{"name":"Grenoble Alpes University Grenoble France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"B\u00e9n\u00e9dicte","family":"Mastroianni","sequence":"additional","affiliation":[{"name":"Department of Medicine Cancer Centre of L\u00e9on B\u00e9rard Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carol","family":"Farver","sequence":"additional","affiliation":[{"name":"Department of Pathology University of Michigan Ann Arbor MI USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fiona","family":"Black","sequence":"additional","affiliation":[{"name":"Department of Cellular Pathology Newcastle upon Tyne Hospitals NHS Foundation Trust Newcastle upon Tyne UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sanjay","family":"Popat","sequence":"additional","affiliation":[{"name":"National Heart and Lung Institute Imperial College London 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