{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,17]],"date-time":"2026-09-17T22:41:43Z","timestamp":1789684903953,"version":"4.0.1"},"reference-count":72,"publisher":"SAGE Publications","issue":"6","license":[{"start":{"date-parts":[[2019,6,1]],"date-time":"2019-06-01T00:00:00Z","timestamp":1559347200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2019,6,1]],"date-time":"2019-06-01T00:00:00Z","timestamp":1559347200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Advances in Wound Care"],"published-print":{"date-parts":[[2019,6]]},"abstract":"<jats:sec>\n                    <jats:title>Significance:<\/jats:title>\n                    <jats:p>Generalized lymphatic anomaly and Gorham\u2013Stout disease are extremely rare diseases with severe symptoms and poor prognosis. The etiology and clinical presentation of the patients remain poorly defined, but recent research has attempted to determine the pathogenesis of these diseases.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Recent Advances:<\/jats:title>\n                    <jats:p>\n                      In recent years, the characteristics of complex lymphatic anomalies have been revealed. Kaposiform lymphangiomatosis is recognized as a new entity that has an aggressive course and poor prognosis. Genetic analysis revealed somatic mutations in genes associated with the phosphoinositide 3-kinase (PI3K) pathway in lymphatic malformation lesions. Somatic\n                      <jats:italic toggle=\"yes\">NRAS<\/jats:italic>\n                      mutation in lymphatic endothelium from a generalized lymphatic anomaly patient was also detected as a potential cause of disease. Furthermore, studies demonstrated the efficacy of the mammalian target of rapamycin (mTOR) inhibitor sirolimus for these lymphatic diseases.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Critical Issues:<\/jats:title>\n                    <jats:p>These diseases have overlapping symptoms, imaging features, and complications, leading to difficulty in their differential diagnosis. In addition, there are no standard therapies. Therefore, we need to determine the differences among these diseases to not only diagnose but also treat them appropriately.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Future Directions:<\/jats:title>\n                    <jats:p>Further investigations should reveal differences in the clinical features and findings of radiological, pathological, and genetic examinations to manage each disease appropriately. Somatic mutation in genes encoding RAS\/PI3K\/mTOR signaling pathway components could be associated with the pathogenesis of these diseases and may be novel targets for drug therapies.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1089\/wound.2018.0850","type":"journal-article","created":{"date-parts":[[2019,1,8]],"date-time":"2019-01-08T00:26:08Z","timestamp":1546907168000},"page":"230-245","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":110,"title":["Generalized Lymphatic Anomaly and Gorham\u2013Stout Disease: Overview and Recent Insights"],"prefix":"10.1177","volume":"8","author":[{"given":"Michio","family":"Ozeki","sequence":"first","affiliation":[{"name":"Department of Pediatrics, Gifu University Graduate School of Medicine, Gifu, Japan."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Toshiyuki","family":"Fukao","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Gifu University Graduate School of Medicine, Gifu, Japan."}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2019,6,6]]},"reference":[{"key":"e_1_3_4_2_2","unstructured":"International Society for the Study of Vascular Anomalies: ISSVA classification for Vascular Anomalies (approved at the May 2018 General Assembly in Amsterdam the Netherlands). http:\/\/issva.org\/classification (last accessed June2018)."},{"key":"e_1_3_4_3_2","doi-asserted-by":"publisher","DOI":"10.1053\/j.sempedsurg.2014.07.006"},{"key":"e_1_3_4_4_2","doi-asserted-by":"publisher","DOI":"10.1089\/lrb.2011.0020"},{"key":"e_1_3_4_5_2","doi-asserted-by":"publisher","DOI":"10.1196\/annals.1413.022"},{"key":"e_1_3_4_6_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.bone.2014.02.011"},{"key":"e_1_3_4_7_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2013.10.013"},{"key":"e_1_3_4_8_2","doi-asserted-by":"publisher","DOI":"10.1513\/AnnalsATS.201308-287BC"},{"key":"e_1_3_4_9_2","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.25278"},{"key":"e_1_3_4_10_2","doi-asserted-by":"publisher","DOI":"10.1164\/ajrccm.161.3.9904056"},{"key":"e_1_3_4_11_2","doi-asserted-by":"publisher","DOI":"10.1038\/nm1689"},{"key":"e_1_3_4_12_2","doi-asserted-by":"publisher","DOI":"10.1161\/CIRCRESAHA.114.303119"},{"key":"e_1_3_4_13_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajpath.2017.05.009"},{"key":"e_1_3_4_14_2","doi-asserted-by":"publisher","DOI":"10.7554\/eLife.34323"},{"key":"e_1_3_4_15_2","doi-asserted-by":"publisher","DOI":"10.1097\/PRS.0000000000004294"},{"key":"e_1_3_4_16_2","doi-asserted-by":"publisher","DOI":"10.1172\/JCI71614"},{"key":"e_1_3_4_17_2","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddu505"},{"key":"e_1_3_4_18_2","doi-asserted-by":"publisher","DOI":"10.1007\/s10456-014-9453-2"},{"key":"e_1_3_4_19_2","doi-asserted-by":"publisher","DOI":"10.1007\/s10456-018-9595-8"},{"key":"e_1_3_4_20_2","doi-asserted-by":"publisher","DOI":"10.1007\/s00256-012-1565-4"},{"key":"e_1_3_4_21_2","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.25914"},{"key":"e_1_3_4_22_2","doi-asserted-by":"publisher","DOI":"10.1053\/j.tvir.2016.10.008"},{"key":"e_1_3_4_23_2","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2014-3673"},{"key":"e_1_3_4_24_2","first-page":"137","article-title":"Vascular Tumours, Vol","author":"Fletcher CDM","year":"2013","unstructured":"Fletcher CDM, , Bridge JA, , Hogendoorn PCW, , Eds FM. 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