Isolated angiokeratomas of the tongue: A rare entity

Angiokeratomas consist of ectasias of dermal capillaries associated with an acanthotic and hyperkeratotic overlying epidermis. These dark red-to-purple, papular vascular anomalies can vary considerably in size, depth, and location. It is a skin disorder that rarely involves oral cavity. It can occur...

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Main Authors: Rizwan Hamid, Altaf H Chalkoo, Inderpreet Singh, Suhail Wani, Sheikh Bilal
Format: Article
Language:English
Published: Wolters Kluwer Medknow Publications 2019-01-01
Series:Indian Journal of Dental Research
Subjects:
Online Access:http://www.ijdr.in/article.asp?issn=0970-9290;year=2019;volume=30;issue=2;spage=322;epage=326;aulast=Hamid
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author Rizwan Hamid
Altaf H Chalkoo
Inderpreet Singh
Suhail Wani
Sheikh Bilal
author_facet Rizwan Hamid
Altaf H Chalkoo
Inderpreet Singh
Suhail Wani
Sheikh Bilal
author_sort Rizwan Hamid
collection DOAJ
description Angiokeratomas consist of ectasias of dermal capillaries associated with an acanthotic and hyperkeratotic overlying epidermis. These dark red-to-purple, papular vascular anomalies can vary considerably in size, depth, and location. It is a skin disorder that rarely involves oral cavity. It can occur in localized or generalized form and is often associated with underlying metabolic disorder such as Fabry's disease and fucosidosis. It has many clinical variants with the same underlying histopathology. Mucosal involvement, including the oral cavity, is occasionally found either as a component of the systemic variety, called angiokeratoma corporis diffusum, or associated with cutaneous lesions in more locations. Isolated oral involvement seems to be rather infrequent, and only eighteen cases have been described in the world literature thus far. Isolated multiple angiokeratomas of tongue without plaque formation have been reported only four times before this. Here, we report a fifth case of isolated multiple angiokeratomas of tongue in a 16-year-old female which was confirmed by immunohistochemical pattern in consonance with a blood vessel origin, with expression of CD31, CD34, and von Willebrand factor. The lesion did not express D2-40 and CD45. No other malformation or metabolic disorder was found in the patient.
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spelling doaj.art-ae2e486bc5bf4aee944bacfe391a3cb92022-12-22T00:35:20ZengWolters Kluwer Medknow PublicationsIndian Journal of Dental Research0970-92901998-36032019-01-0130232232610.4103/ijdr.IJDR_644_17Isolated angiokeratomas of the tongue: A rare entityRizwan HamidAltaf H ChalkooInderpreet SinghSuhail WaniSheikh BilalAngiokeratomas consist of ectasias of dermal capillaries associated with an acanthotic and hyperkeratotic overlying epidermis. These dark red-to-purple, papular vascular anomalies can vary considerably in size, depth, and location. It is a skin disorder that rarely involves oral cavity. It can occur in localized or generalized form and is often associated with underlying metabolic disorder such as Fabry's disease and fucosidosis. It has many clinical variants with the same underlying histopathology. Mucosal involvement, including the oral cavity, is occasionally found either as a component of the systemic variety, called angiokeratoma corporis diffusum, or associated with cutaneous lesions in more locations. Isolated oral involvement seems to be rather infrequent, and only eighteen cases have been described in the world literature thus far. Isolated multiple angiokeratomas of tongue without plaque formation have been reported only four times before this. Here, we report a fifth case of isolated multiple angiokeratomas of tongue in a 16-year-old female which was confirmed by immunohistochemical pattern in consonance with a blood vessel origin, with expression of CD31, CD34, and von Willebrand factor. The lesion did not express D2-40 and CD45. No other malformation or metabolic disorder was found in the patient.http://www.ijdr.in/article.asp?issn=0970-9290;year=2019;volume=30;issue=2;spage=322;epage=326;aulast=HamidAngiokeratomaimmuno-histochemicalisolatedoral cavitytonguetumor
spellingShingle Rizwan Hamid
Altaf H Chalkoo
Inderpreet Singh
Suhail Wani
Sheikh Bilal
Isolated angiokeratomas of the tongue: A rare entity
Indian Journal of Dental Research
Angiokeratoma
immuno-histochemical
isolated
oral cavity
tongue
tumor
title Isolated angiokeratomas of the tongue: A rare entity
title_full Isolated angiokeratomas of the tongue: A rare entity
title_fullStr Isolated angiokeratomas of the tongue: A rare entity
title_full_unstemmed Isolated angiokeratomas of the tongue: A rare entity
title_short Isolated angiokeratomas of the tongue: A rare entity
title_sort isolated angiokeratomas of the tongue a rare entity
topic Angiokeratoma
immuno-histochemical
isolated
oral cavity
tongue
tumor
url http://www.ijdr.in/article.asp?issn=0970-9290;year=2019;volume=30;issue=2;spage=322;epage=326;aulast=Hamid
work_keys_str_mv AT rizwanhamid isolatedangiokeratomasofthetonguearareentity
AT altafhchalkoo isolatedangiokeratomasofthetonguearareentity
AT inderpreetsingh isolatedangiokeratomasofthetonguearareentity
AT suhailwani isolatedangiokeratomasofthetonguearareentity
AT sheikhbilal isolatedangiokeratomasofthetonguearareentity