{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,20]],"date-time":"2026-10-20T02:38:02Z","timestamp":1792463882950,"version":"4.5.0"},"reference-count":76,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2025,9,3]],"date-time":"2025-09-03T00:00:00Z","timestamp":1756857600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Surg."],"abstract":"<jats:p>Klippel\u2013Tr\u00e9naunay syndrome (KTS) typically involves a combination of capillary, lymphatic, and venous malformations of the lower extremities. Genitourinary involvement is frequently observed in patients with KTS. Bleeding and pelvic pain are the most common complications. This condition has been increasingly reported in recent years. However, most authors have only depicted extreme presentations or various combinations of clinical findings. The underlying pathophysiology of genitourinary involvement in KTS remains unclear. Genitourinary involvement in female patients with KTS has a more complicated pathophysiology than that in male patients with KTS. After understanding its pathophysiology, some patients can be successfully managed by using a staged plan. Therefore, recognizing the pathophysiology of genitourinary involvement is necessary for practitioners to evaluate and determine adequate management. Owing to the complexity and rarity of this condition, a prospective controlled study involving a large cohort of patients is impossible. Based on a literature review and our practice, we discuss the pathophysiology, evaluation, and treatment strategies for genitourinary involvement in KTS.<\/jats:p>","DOI":"10.3389\/fsurg.2025.1532509","type":"journal-article","created":{"date-parts":[[2025,9,3]],"date-time":"2025-09-03T15:48:15Z","timestamp":1756914495000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":2,"title":["Genitourinary involvement in Klippel\u2013Tr\u00e9naunay syndrome: pathophysiology, evaluation, and management"],"prefix":"10.3389","volume":"12","author":[{"given":"Weilong","family":"Lin","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chong","family":"Xie","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Huaijie","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Weijia","family":"Yang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peihua","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhengtuan","family":"Guo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2025,9,3]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","first-page":"288","DOI":"10.1186\/s13023-023-02857-5","article-title":"Gastrointestinal involvement in Klippel-Tr\u00e9naunay syndrome: pathophysiology, evaluation, and management","volume":"18","author":"Wang","year":"2023","journal-title":"Orphanet J Rare Dis"},{"key":"B2","doi-asserted-by":"publisher","first-page":"e21472","DOI":"10.7759\/cureus.21472","article-title":"Internal iliac vein reflux: an underrecognized pathophysiology in Klippel-Tr\u00e9naunay syndrome with pelvis involvement","volume":"14","author":"Wang","year":"2022","journal-title":"Cureus"},{"key":"B3","doi-asserted-by":"publisher","first-page":"417","DOI":"10.1016\/s0022-3468(84)80266-3","article-title":"Prognosis and management of lesions of the trunk in children with Klippel-Tr\u00e9naunay syndrome","volume":"19","author":"Telander","year":"1984","journal-title":"J Pediatr Surg"},{"key":"B4","doi-asserted-by":"publisher","first-page":"1244","DOI":"10.1016\/j.juro.2006.11.099","article-title":"Klippel-Tr\u00e9naunay syndrome: incidence and treatment of genitourinary sequelae","volume":"177","author":"Husmann","year":"2007","journal-title":"J Urol"},{"key":"B5","doi-asserted-by":"publisher","first-page":"137","DOI":"10.1007\/s11255-005-3154-0","article-title":"Life-threatening gross hematuria due to genitourinary manifestation of Klippel-Tr\u00e9naunay syndrome","volume":"38","author":"Rubenwolf","year":"2006","journal-title":"Int Urol Nephrol"},{"key":"B6","doi-asserted-by":"publisher","first-page":"226","DOI":"10.1159\/000030537","article-title":"Massive hematuria in adults with Klippel-Tr\u00e9naunay syndrome associated with vascular malformation of the bladder","volume":"64","author":"Shekarriz","year":"2000","journal-title":"Urol Int"},{"key":"B7","doi-asserted-by":"publisher","first-page":"e246601","DOI":"10.1136\/bcr-2021-246601","article-title":"Management of postpartum haemorrhage in a patient with Klippel-Tr\u00e9naunay syndrome","volume":"15","author":"Benson","year":"2022","journal-title":"BMJ Case Rep"},{"key":"B8","doi-asserted-by":"publisher","first-page":"1780","DOI":"10.1111\/1471-0528.14698","article-title":"Complications of pregnancy and labour in women with Klippel-Tr\u00e9naunay syndrome: a nationwide cross-sectional study","volume":"124","author":"Horbach","year":"2017","journal-title":"BJOG"},{"key":"B9","doi-asserted-by":"publisher","first-page":"2783","DOI":"10.1111\/j.1572-0241.2001.04110.x","article-title":"Bleeding from cavernous angiomatosis of the rectum in Klippel-Tr\u00e9naunay syndrome: report of three cases and literature review","volume":"96","author":"Wilson","year":"2001","journal-title":"Am J Gastroenterol"},{"key":"B10","doi-asserted-by":"publisher","first-page":"291","DOI":"10.1177\/026835550702200611","article-title":"Klippel-Tr\u00e9naunay syndrome: current management","volume":"22","author":"Gloviczki","year":"2007","journal-title":"Phlebology"},{"key":"B11","doi-asserted-by":"publisher","first-page":"353","DOI":"10.1097\/00000658-198303000-00017","article-title":"Surgical implications of Klippel-Tr\u00e9naunay syndrome","volume":"197","author":"Gloviczki","year":"1983","journal-title":"Ann Surg"},{"key":"B12","doi-asserted-by":"publisher","first-page":"893320","DOI":"10.3389\/fsurg.2022.893320","article-title":"Uterine involvement in Klippel-Tr\u00e9naunay syndrome: a rare but relevant event. 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