{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,4]],"date-time":"2026-09-04T21:26:29Z","timestamp":1788557189361,"version":"build-2803163510"},"reference-count":13,"publisher":"BMJ","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Reports"],"accepted":{"date-parts":[[2014,9,26]]},"abstract":"<jats:p>Chyloptysis and chylous pulmonary congestion are extremely rare complications of lymphangioleiomyomatosis (LAM). We report a case of a 50-year-old woman with tuberous sclerosis complex-associated LAM, who presented with expectorating milky-white bronchial casts. She was diagnosed with chyloptysis and chylous pulmonary congestion by sputum analysis. Her symptoms and lung infiltration were improved by oral sirolimus therapy; moreover, serum Krebs von den Lungen-6 (KL-6) levels paralleled the symptoms and lung infiltration of these complications. We suggest that serum KL-6 may be a useful monitoring biomarker of chyloptysis and chylous pulmonary congestion in LAM.<\/jats:p>","DOI":"10.1136\/bcr-2014-205701","type":"journal-article","created":{"date-parts":[[2014,10,17]],"date-time":"2014-10-17T08:20:19Z","timestamp":1413534019000},"page":"bcr2014205701","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":3,"title":["Recovery of chylous pulmonary congestion in tuberous sclerosis complex-associated lymphangioleiomyomatosis"],"prefix":"10.1136","volume":"2014","author":[{"given":"Yoshiaki","family":"Kinoshita","sequence":"first","affiliation":[{"name":"Division of Respiratory Medicine, Department of Internal Medicine, National Hospital Organization, Kokura Medical Center, Kitakyushu, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Atsuhiko","family":"Sakamoto","sequence":"additional","affiliation":[{"name":"Division of Respiratory Medicine, Department of Internal Medicine, National Hospital Organization, Kokura Medical Center, Kitakyushu, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kouko","family":"Hidaka","sequence":"additional","affiliation":[{"name":"Division of Respiratory Medicine, Department of Internal Medicine, National Hospital Organization, Kokura Medical Center, Kitakyushu, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"239","published-online":{"date-parts":[[2014,10,17]]},"reference":[{"key":"2025082802385539000_2014.oct17_1.bcr2014205701.1","doi-asserted-by":"publisher","DOI":"10.1378\/chest.07-0898"},{"key":"2025082802385539000_2014.oct17_1.bcr2014205701.2","doi-asserted-by":"publisher","DOI":"10.1378\/chest.125.1.336"},{"key":"2025082802385539000_2014.oct17_1.bcr2014205701.3","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa063564"},{"key":"2025082802385539000_2014.oct17_1.bcr2014205701.4","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa1100391"},{"key":"2025082802385539000_2014.oct17_1.bcr2014205701.5","doi-asserted-by":"publisher","DOI":"10.7326\/0003-4819-154-12-201106210-00007"},{"key":"2025082802385539000_2014.oct17_1.bcr2014205701.6","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMc086040"},{"key":"2025082802385539000_2014.oct17_1.bcr2014205701.7","doi-asserted-by":"publisher","DOI":"10.1164\/ajrccm.186.4.389"},{"key":"2025082802385539000_2014.oct17_1.bcr2014205701.8","doi-asserted-by":"publisher","DOI":"10.1016\/j.resinv.2013.03.002"},{"key":"2025082802385539000_2014.oct17_1.bcr2014205701.9","doi-asserted-by":"publisher","DOI":"10.1164\/ajrccm.156.1.9612043"},{"key":"2025082802385539000_2014.oct17_1.bcr2014205701.10","doi-asserted-by":"publisher","DOI":"10.1164\/ajrccm\/148.3.637"},{"key":"2025082802385539000_2014.oct17_1.bcr2014205701.11","first-page":"A2024","article-title":"Diagnostic value of simultaneous measurements of serum KL-6 and SP-A\/D in multifocal micronodular pneumocyte hyperplasia (mmph)","volume":"187","author":"Ogi","year":"2013","journal-title":"Am J Respir Crit Care Med"},{"key":"2025082802385539000_2014.oct17_1.bcr2014205701.12","doi-asserted-by":"publisher","DOI":"10.1080\/00313020802198077"},{"key":"2025082802385539000_2014.oct17_1.bcr2014205701.13","doi-asserted-by":"publisher","DOI":"10.1186\/1471-2466-11-22"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2014-205701","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,28]],"date-time":"2025-08-28T09:39:03Z","timestamp":1756373943000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2014-205701"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2014,10,17]]},"references-count":13,"alternative-id":["10.1136\/bcr-2014-205701"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2014-205701","relation":{},"ISSN":["1757-790X"],"issn-type":[{"value":"1757-790X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2014,10,17]]}}}