{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,13]],"date-time":"2026-03-13T12:35:53Z","timestamp":1773405353415,"version":"3.50.1"},"reference-count":17,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2016,11,16]],"date-time":"2016-11-16T00:00:00Z","timestamp":1479254400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J Cutan Pathol"],"published-print":{"date-parts":[[2017,1]]},"abstract":"<jats:p>Retiform hemangioendothelioma (<jats:styled-content style=\"fixed-case\">RH<\/jats:styled-content>) is a rare vascular neoplasm with a high rate of local recurrence and low metastatic potential. We describe an unusual case of <jats:styled-content style=\"fixed-case\">RH<\/jats:styled-content> in a 45\u2010year\u2010old patient with Milroy disease, with a prominent solid component diffusely involving a chronic lymphedematous leg. This case is consistent with the postulated relationship between lymphedema and vascular neoplasms developing as a result of local immune dysfunction, and highlights the need to closely monitor patients with Milroy disease for pathologic changes. Our case highlights a unique example of <jats:styled-content style=\"fixed-case\">RH<\/jats:styled-content> with atypical features. There are several noteworthy unusual clinical and histologic findings including diffuse involvement of an entire limb, solid component with cytologic atypia, <jats:styled-content style=\"fixed-case\">D2<\/jats:styled-content>\u201040 expression, and first\u2010time\u2010reported association with Milroy disease. Given the atypical histologic presentation of cytologic atypia, solid areas and atypical immunohistochemical profile with <jats:styled-content style=\"fixed-case\">D2<\/jats:styled-content>\u201040 positivity, this case could cause diagnostic difficulty, especially in the setting of such a broad clinical differential.<\/jats:p>","DOI":"10.1111\/cup.12844","type":"journal-article","created":{"date-parts":[[2016,10,13]],"date-time":"2016-10-13T17:20:52Z","timestamp":1476379252000},"page":"98-103","source":"Crossref","is-referenced-by-count":5,"title":["Atypical retiform hemangioendothelioma arising in a patient with Milroy disease: a case report and review of the literature"],"prefix":"10.1111","volume":"44","author":[{"given":"Aileen Grace P.","family":"Arriola","sequence":"first","affiliation":[{"name":"Pathology and Laboratory Medicine Hospital of the University of Pennsylvania  Philadelphia PA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Laura 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