{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,2,21]],"date-time":"2025-02-21T19:24:43Z","timestamp":1740165883866,"version":"3.37.3"},"reference-count":13,"publisher":"Springer Science and Business Media LLC","issue":"2","license":[{"start":{"date-parts":[[2022,11,11]],"date-time":"2022-11-11T00:00:00Z","timestamp":1668124800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.springernature.com\/gp\/researchers\/text-and-data-mining"},{"start":{"date-parts":[[2022,11,11]],"date-time":"2022-11-11T00:00:00Z","timestamp":1668124800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/www.springernature.com\/gp\/researchers\/text-and-data-mining"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["CEN Case Rep"],"DOI":"10.1007\/s13730-022-00747-6","type":"journal-article","created":{"date-parts":[[2022,11,11]],"date-time":"2022-11-11T18:02:55Z","timestamp":1668189775000},"page":"205-209","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["A case of chyluria with nephrotic-range proteinuria caused by lymphatic malformation, leading to a diagnosis of Klippel\u2013Trenaunay syndrome"],"prefix":"10.1007","volume":"12","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0827-9619","authenticated-orcid":false,"given":"Yuichiro","family":"Izumi","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ryosuke","family":"Date","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Teruhiko","family":"Mizumoto","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Terumasa","family":"Nakagawa","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yutaka","family":"Kakizoe","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Masataka","family":"Adachi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takashige","family":"Kuwabara","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Masashi","family":"Mukoyama","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2022,11,11]]},"reference":[{"key":"747_CR1","doi-asserted-by":"publisher","first-page":"c248","DOI":"10.1159\/000329154","volume":"119","author":"G Graziani","year":"2011","unstructured":"Graziani G, Cucchiari D, Verdesca S, Balzarini L, Montanelli A, Ponticelli C. Chyluria associated with nephrotic-range proteinuria: pathophysiology, clinical picture and therapeutic options. Nephron Clin Pract. 2011;119:c248\u201354.","journal-title":"Nephron Clin Pract"},{"key":"747_CR2","doi-asserted-by":"publisher","first-page":"28","DOI":"10.1016\/S0025-6196(11)63615-X","volume":"73","author":"AG Jacob","year":"1998","unstructured":"Jacob AG, Driscoll DJ, Shaughnessy WJ, Stanson AW, Clay RP, Gloviczki P. Klippel-Trenaunay syndrome: spectrum and management. Mayo Clin Proc. 1998;73:28\u201336.","journal-title":"Mayo Clin Proc"},{"key":"747_CR3","doi-asserted-by":"publisher","first-page":"201","DOI":"10.2147\/VHRM.S358849","volume":"18","author":"P Harnarayan","year":"2022","unstructured":"Harnarayan P, Harnanan D. The Klippel-Trenaunay syndrome in 2022: unraveling its genetic and molecular profile and its link to the limb overgrowth syndromes. Vasc Health Risk Manag. 2022;18:201\u20139.","journal-title":"Vasc Health Risk Manag"},{"key":"747_CR4","doi-asserted-by":"publisher","first-page":"685","DOI":"10.1067\/S0022-3476(03)00288-9","volume":"143","author":"K Kano","year":"2003","unstructured":"Kano K, Arisaka O. Chyluria due to retroperitoneal lymphangioma producing nephrotic syndorome. J Pediatr. 2003;143:685.","journal-title":"J Pediatr"},{"key":"747_CR5","doi-asserted-by":"publisher","first-page":"303","DOI":"10.1097\/00000658-196508000-00023","volume":"162","author":"SM Lindenauer","year":"1965","unstructured":"Lindenauer SM. The Klippel-Trenaunay syndrome: varicosity, hypertrophy and hemangioma with no arteriovenous fistula. Ann Surg. 1965;162:303\u201314.","journal-title":"Ann Surg"},{"key":"747_CR6","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0123092","volume":"10","author":"DC Loconte","year":"2015","unstructured":"Loconte DC, Grossi V, Bozzao C, Forte G, Bagnulo R, Stella A, et al. Molecular and functional characterization of three different postzygotic mutations in PIK3CA-related overgrowth spectrum (PROS) patients: effects on PI3K\/AKT\/mTOR signaling and sensitivity to PIK3 inhibitors. PLoS ONE. 2015;10: e0123092.","journal-title":"PLoS ONE"},{"key":"747_CR7","doi-asserted-by":"publisher","first-page":"492","DOI":"10.1002\/ajmg.1320590416","volume":"59","author":"AJ Whelan","year":"1995","unstructured":"Whelan AJ, Watson MS, Porter FD, Steiner RD. Klippel-Trenaunay-Weber syndrome associated with a 5:11 balanced translocation. Am J Med Genet. 1995;59:492\u20134.","journal-title":"Am J Med Genet"},{"key":"747_CR8","doi-asserted-by":"publisher","first-page":"640","DOI":"10.1038\/nature02320","volume":"427","author":"XL Tian","year":"2004","unstructured":"Tian XL, Kadaba R, You SA, Liu M, Timur AA, Yang L, et al. Identification of an angiogenic factor that when mutated causes susceptibility to Klippel-Trenaunay syndrome. Nature. 2004;427:640\u20135.","journal-title":"Nature"},{"key":"747_CR9","doi-asserted-by":"publisher","first-page":"183","DOI":"10.1159\/000059343","volume":"95","author":"Q Wang","year":"2001","unstructured":"Wang Q, Timur AA, Szafranski P, Sadgephour A, Jurecic V, Cowell J, et al. Identification and molecular characterization of de novo translocation t(8;14)(q22.3;q13) associated with a vascular and tissue overgrowth syndrome. Cytogenet Cell Genet. 2001;95:183\u20138.","journal-title":"Cytogenet Cell Genet"},{"key":"747_CR10","doi-asserted-by":"publisher","first-page":"1048","DOI":"10.1016\/j.jpeds.2014.12.069","volume":"166","author":"VL Luks","year":"2015","unstructured":"Luks VL, Kamitaki N, Vivero MP, Uller W, Rab R, Bovee JVMG, et al. Lymphatic and other vascular malformative\/overgrowth disorders are caused by somatic mutations in PIK3CA. J Pediatr. 2015;166:1048\u201354.","journal-title":"J Pediatr"},{"key":"747_CR11","doi-asserted-by":"publisher","first-page":"17","DOI":"10.1111\/exd.12826","volume":"25","author":"H Vahidnezhad","year":"2016","unstructured":"Vahidnezhad H, Youssefian L, Uitto J. Klippel-Trenaunay syndrome belongs to the PIK3CA-related overgrowth spectrum (PROS). Exp Dermatol. 2016;25:17\u20139.","journal-title":"Exp Dermatol"},{"key":"747_CR12","doi-asserted-by":"publisher","first-page":"540","DOI":"10.1038\/s41586-018-0217-9","volume":"558","author":"Q Venot","year":"2018","unstructured":"Venot Q, Blanc T, Rabia SH, Berteloot L, Ladraa S, Duong JP, et al. Targeted therapy in patients with PIK3CA-related overgrowth syndrome. Nature. 2018;558:540\u20136.","journal-title":"Nature"},{"key":"747_CR13","doi-asserted-by":"publisher","first-page":"2433","DOI":"10.1038\/s41436-021-01290-y","volume":"23","author":"M Luu","year":"2021","unstructured":"Luu M, Vabres P, Devilliers H, Loffroy R, Phan A, Martin L, et al. Safety and efficacy of low-dose PI3K inhibitor taselisib in adult patients with CLOVES and Klippel-Trenaunay syndrome (KTS): the TOTEM trial, a phase 1\/2 multicenter, open-label, single-arm study. Genet Med. 2021;23:2433\u201342.","journal-title":"Genet Med"}],"container-title":["CEN Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s13730-022-00747-6.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1007\/s13730-022-00747-6\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s13730-022-00747-6.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,5,1]],"date-time":"2023-05-01T07:10:28Z","timestamp":1682925028000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/s13730-022-00747-6"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,11,11]]},"references-count":13,"journal-issue":{"issue":"2","published-online":{"date-parts":[[2023,5]]}},"alternative-id":["747"],"URL":"https:\/\/doi.org\/10.1007\/s13730-022-00747-6","relation":{},"ISSN":["2192-4449"],"issn-type":[{"type":"electronic","value":"2192-4449"}],"subject":[],"published":{"date-parts":[[2022,11,11]]},"assertion":[{"value":"9 August 2022","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"6 November 2022","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"11 November 2022","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"The authors have declared that no conflict of interest exists.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}},{"value":"The patients provided written informed consent to publish this case report, including case description, medical data, and images, maintaining anonymity.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}}]}}