Central Nervous System Xanthoma Disseminatum: Response to 2CdA in an Adolescent

Xanthoma disseminatum is a normolipemic non-Langerhans cell histiocytosis characterized by red-brown rubbery papules of the skin which coalesce into plaque-like lesions with symmetric involvement of face, flexor, and intertriginous areas. Less commonly, xanthoma disseminatum may affect mucosal linin...

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Main Authors: Patrick DeMoss, Nancy Tang, Kristen Yeom, Audris Chiang, Ann L. Marqueling, Michael R. Jeng
Format: Article
Language:English
Published: Wiley 2022-01-01
Series:Case Reports in Pediatrics
Online Access:http://dx.doi.org/10.1155/2022/9906668
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author Patrick DeMoss
Nancy Tang
Kristen Yeom
Audris Chiang
Ann L. Marqueling
Michael R. Jeng
author_facet Patrick DeMoss
Nancy Tang
Kristen Yeom
Audris Chiang
Ann L. Marqueling
Michael R. Jeng
author_sort Patrick DeMoss
collection DOAJ
description Xanthoma disseminatum is a normolipemic non-Langerhans cell histiocytosis characterized by red-brown rubbery papules of the skin which coalesce into plaque-like lesions with symmetric involvement of face, flexor, and intertriginous areas. Less commonly, xanthoma disseminatum may affect mucosal linings, abdominal organs, and the central nervous system, leading to endocrinopathies. We report a 12-year-old adolescent with mucosal, central nervous system, and painful cutaneous lesions, further complicated by diabetes insipidus and amenorrhea. Treatment with 2-chlorodeoxyadenosine led to relief of pain and significant improvement of mucosal, central nervous system, and cutaneous lesions, with subsequent restoration of menstrual cycles.
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series Case Reports in Pediatrics
spelling doaj-art-07a2d46db69e4a85a3102a5408c51bc82025-08-20T02:01:35ZengWileyCase Reports in Pediatrics2090-68112022-01-01202210.1155/2022/9906668Central Nervous System Xanthoma Disseminatum: Response to 2CdA in an AdolescentPatrick DeMoss0Nancy Tang1Kristen Yeom2Audris Chiang3Ann L. Marqueling4Michael R. Jeng5Departments of Pediatrics (PDDepartments of Pediatrics (PDDepartments of Pediatrics (PDDepartments of Pediatrics (PDDepartments of Pediatrics (PDDepartments of Pediatrics (PDXanthoma disseminatum is a normolipemic non-Langerhans cell histiocytosis characterized by red-brown rubbery papules of the skin which coalesce into plaque-like lesions with symmetric involvement of face, flexor, and intertriginous areas. Less commonly, xanthoma disseminatum may affect mucosal linings, abdominal organs, and the central nervous system, leading to endocrinopathies. We report a 12-year-old adolescent with mucosal, central nervous system, and painful cutaneous lesions, further complicated by diabetes insipidus and amenorrhea. Treatment with 2-chlorodeoxyadenosine led to relief of pain and significant improvement of mucosal, central nervous system, and cutaneous lesions, with subsequent restoration of menstrual cycles.http://dx.doi.org/10.1155/2022/9906668
spellingShingle Patrick DeMoss
Nancy Tang
Kristen Yeom
Audris Chiang
Ann L. Marqueling
Michael R. Jeng
Central Nervous System Xanthoma Disseminatum: Response to 2CdA in an Adolescent
Case Reports in Pediatrics
title Central Nervous System Xanthoma Disseminatum: Response to 2CdA in an Adolescent
title_full Central Nervous System Xanthoma Disseminatum: Response to 2CdA in an Adolescent
title_fullStr Central Nervous System Xanthoma Disseminatum: Response to 2CdA in an Adolescent
title_full_unstemmed Central Nervous System Xanthoma Disseminatum: Response to 2CdA in an Adolescent
title_short Central Nervous System Xanthoma Disseminatum: Response to 2CdA in an Adolescent
title_sort central nervous system xanthoma disseminatum response to 2cda in an adolescent
url http://dx.doi.org/10.1155/2022/9906668
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