{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,11]],"date-time":"2026-02-11T04:23:09Z","timestamp":1770783789446,"version":"3.50.0"},"reference-count":20,"publisher":"Archives of Pathology and Laboratory Medicine","issue":"9","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2000,9]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>A case of congenital lingual angiodysplasia with macroglossia in a 5-year-old girl is presented. A diffusely enlarged tongue was present at birth and continued to grow as the child aged. It was accompanied by impaired speech, difficulty in eating and breathing, and sleep apnea, necessitating surgical intervention. The fundamental lesion represents a complex vascular malformation of the lymphangioma-hemangioma type, involving extensively the deep musculature of the tongue. Multifocal and multicentric cavernous lymphangioma-like and hemangioma-like areas merge with benign angioendotheliomatous-like foci in a background of variable muscle degeneration and marked fibrosis. Neither a borderline nor an overtly malignant vasoformative neoplasm was present. Because of its distinctively widespread, multicentric intramuscular distribution, this lesion may be construed as a diffuse variant of lingual lymphangioma-hemangioma malformation, closely resembling a previously described case of macroglossal lymphangioendotheliomatosis.<\/jats:p>","DOI":"10.5858\/2000-124-1349-cmal","type":"journal-article","created":{"date-parts":[[2021,1,28]],"date-time":"2021-01-28T21:45:06Z","timestamp":1611870306000},"page":"1349-1351","source":"Crossref","is-referenced-by-count":0,"title":["Congenital Macroglossal Angiodysplasia (\u201cLymphangioendotheliomatosis\u201d)"],"prefix":"10.5858","volume":"124","author":[{"given":"Ghazi","family":"Bakaeen","sequence":"first","affiliation":[{"name":"From the Faculty of Dentistry, University of Jordan, Amman, Jordan (Drs G. Bakaeen and Rehani); Department of Restorative Dentistry, Temple University School of Dentistry, Philadelphia, Pa (Drs Winkler and L. Bakaeen); Department of Pediatrics, MCP Hahnemann University, and Section of Neurology and Department of Pathology and Laboratory Medicine, St Christopher's Hospital for Children, Philadelphia, Pa (Dr Katsetos)."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sheldon","family":"Winkler","sequence":"additional","affiliation":[{"name":"Reprints: Sheldon Winkler, DDS, Department of Restorative Dentistry, Temple University School of Dentistry, 3223 N Broad St, Philadelphia, PA 19140-5096."},{"name":"From the Faculty of Dentistry, University of Jordan, Amman, Jordan (Drs G. Bakaeen and Rehani); Department of Restorative Dentistry, Temple University School of Dentistry, Philadelphia, Pa (Drs Winkler and L. Bakaeen); Department of Pediatrics, MCP Hahnemann University, and Section of Neurology and Department of Pathology and Laboratory Medicine, St Christopher's Hospital for Children, Philadelphia, Pa (Dr Katsetos)."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lara","family":"Bakaeen","sequence":"additional","affiliation":[{"name":"From the Faculty of Dentistry, University of Jordan, Amman, Jordan (Drs G. Bakaeen and Rehani); Department of Restorative Dentistry, Temple University School of Dentistry, Philadelphia, Pa (Drs Winkler and L. Bakaeen); Department of Pediatrics, MCP Hahnemann University, and Section of Neurology and Department of Pathology and Laboratory Medicine, St Christopher's Hospital for Children, Philadelphia, Pa (Dr Katsetos)."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lina A.","family":"Rehani","sequence":"additional","affiliation":[{"name":"From the Faculty of Dentistry, University of Jordan, Amman, Jordan (Drs G. Bakaeen and Rehani); Department of Restorative Dentistry, Temple University School of Dentistry, Philadelphia, Pa (Drs Winkler and L. Bakaeen); Department of Pediatrics, MCP Hahnemann University, and Section of Neurology and Department of Pathology and Laboratory Medicine, St Christopher's Hospital for Children, Philadelphia, Pa (Dr Katsetos)."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Christos D.","family":"Katsetos","sequence":"additional","affiliation":[{"name":"From the Faculty of Dentistry, University of Jordan, Amman, Jordan (Drs G. Bakaeen and Rehani); Department of Restorative Dentistry, Temple University School of Dentistry, Philadelphia, Pa (Drs Winkler and L. Bakaeen); Department of Pediatrics, MCP Hahnemann University, and Section of Neurology and Department of Pathology and Laboratory Medicine, St Christopher's Hospital for Children, Philadelphia, Pa (Dr Katsetos)."}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"3906","reference":[{"key":"i1543-2165-124-9-1349-b1","series-title":"Head Neck Surg","first-page":"231","article-title":"The pathology of head and neck tumors: vasoformative tumors, part 9A.","volume":"3","author":"Batsakis, J. G.","year":"1981","unstructured":"Batsakis, J. G. and D. H.Rice. The pathology of head and neck tumors: vasoformative tumors, part 9A.Head Neck Surg1981. 3:231\u2013239."},{"key":"i1543-2165-124-9-1349-b2","series-title":"Am J Otolaryngol","first-page":"367","article-title":"Noncutaneous cavernous hemangiomas of the head and neck.","volume":"18","author":"Shpitzer, T.","year":"1997","unstructured":"Shpitzer, T., A. M.Noyek, and I.Witterick. et al. Noncutaneous cavernous hemangiomas of the head and neck.Am J Otolaryngol1997. 18:367\u2013374."},{"key":"i1543-2165-124-9-1349-b3","series-title":"Br J Oral Maxillofac Surg","first-page":"92","article-title":"Oral and maxillofacial tumours in children: a review.","volume":"35","author":"Sato, M.","year":"1997","unstructured":"Sato, M., N.Tanaka, T.Sato, and T.Amagasa. Oral and maxillofacial tumours in children: a review.Br J Oral Maxillofac Surg1997. 35:92\u201395."},{"key":"i1543-2165-124-9-1349-b4","series-title":"Arch Otolaryngol","first-page":"600","article-title":"Macroglossal lymphangioendotheliomatosis: report of a case in a pregnant woman.","volume":"110","author":"Loeffler, J. R.","year":"1984","unstructured":"Loeffler, J. R., P. H.Duray, and C. T.Sasaki. Macroglossal lymphangioendotheliomatosis: report of a case in a pregnant woman.Arch Otolaryngol1984. 110:600\u2013603."},{"key":"i1543-2165-124-9-1349-b5","series-title":"Am J Pathol","first-page":"469","article-title":"Cellular distribution of retinoic acid receptor-\u03b1 (RAR\u03b1) protein in serous adenocarcinomas of ovarian, tubal and peritoneal origin: comparison with estrogen receptor status.","volume":"153","author":"Katsetos, C. D.","year":"1998","unstructured":"Katsetos, C. D., I.Stadnicka, and J. C.Boyd. et al. Cellular distribution of retinoic acid receptor-\u03b1 (RAR\u03b1) protein in serous adenocarcinomas of ovarian, tubal and peritoneal origin: comparison with estrogen receptor status.Am J Pathol1998. 153:469\u2013480."},{"key":"i1543-2165-124-9-1349-b6","series-title":"J Oral Maxillofac Surg","first-page":"932","article-title":"Lymphangiomas of the oral cavity: a clinicopathologic, immunohistochemical, and electron-microscopic study.","volume":"55","author":"Brennan, T. D.","year":"1997","unstructured":"Brennan, T. D., A. S.Miller, and S. Y.Chen. 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