Xanthoma disseminatum with extensive respiratory involvement effectively treated with cladribine: a case report

Xanthoma disseminatum (XD) is a rare and benign proliferative systemic disease that usually affects the skin and mucosal membranes with variable extent. Extensive systemic involvement can be associated with higher morbidity. There is paucity in the literature describing this rare pathological entity...

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Autores principales:	Heba Al-Tarcheh, Shahed Tish, Salloum Salloum, Ahed Haj Ibrahim
Formato:	article
Lenguaje:	EN
Publicado:	Thieme Medical and Scientific Publishers Pvt. Ltd. 2020
Materias:	cladribine histiocytosis non-langerhans Medicine R
Acceso en línea:	https://doaj.org/article/7d7f6313f38240c98387ccb037dab948
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spelling	oai:doaj.org-article:7d7f6313f38240c98387ccb037dab9482021-12-02T17:00:53ZXanthoma disseminatum with extensive respiratory involvement effectively treated with cladribine: a case report2231-07702249-446410.4103/ajm.ajm_177_19https://doaj.org/article/7d7f6313f38240c98387ccb037dab9482020-04-01T00:00:00Zhttp://www.thieme-connect.de/DOI/DOI?10.4103/ajm.ajm_177_19https://doaj.org/toc/2231-0770https://doaj.org/toc/2249-4464Xanthoma disseminatum (XD) is a rare and benign proliferative systemic disease that usually affects the skin and mucosal membranes with variable extent. Extensive systemic involvement can be associated with higher morbidity. There is paucity in the literature describing this rare pathological entity, and the ideal management remains controversial. In this article, we report our experience with cladribine in treating a case of XD. We documented the clinical and pathological manifestations of a 24-year-old woman who was initially diagnosed with rheumatoid arthritis. She presented to our institute with respiratory compromise and was found to have XD affecting skin, mucosal membranes, joints, and bone marrow. The patient received six cycles of cladribine for 6 months, during which she showed a remarkable response in relation to the respiratory lesions. Her hemoglobin also normalized and inflammatory markers gradually decreased to reach normal values. However, her skin lesions did not respond to treatment but no new lesions appeared. With our experience with cladribine, we believe that it could be a promising treatment option for XD. However, more work has to be conducted to determine the efficacy and safety in the long term. Key message: Given the rarity of this understudied entity, the natural history and the ultimate treatment remain unclear. We highlight the natural history and clinical course of xanthoma disseminatum (XD) in this article. We also describe our experience with cladribine in treating XD. We believe that similar experiences should be compiled to better understand this pathology and the effective therapy options for these patients.Heba Al-TarchehShahed TishSalloum SalloumAhed Haj IbrahimThieme Medical and Scientific Publishers Pvt. Ltd.articlecladribinehistiocytosisnon-langerhansMedicineRENAvicenna Journal of Medicine, Vol 10, Iss 02, Pp 83-88 (2020)
institution	DOAJ
collection	DOAJ
language	EN
topic	cladribine histiocytosis non-langerhans Medicine R
spellingShingle	cladribine histiocytosis non-langerhans Medicine R Heba Al-Tarcheh Shahed Tish Salloum Salloum Ahed Haj Ibrahim Xanthoma disseminatum with extensive respiratory involvement effectively treated with cladribine: a case report
description	Xanthoma disseminatum (XD) is a rare and benign proliferative systemic disease that usually affects the skin and mucosal membranes with variable extent. Extensive systemic involvement can be associated with higher morbidity. There is paucity in the literature describing this rare pathological entity, and the ideal management remains controversial. In this article, we report our experience with cladribine in treating a case of XD. We documented the clinical and pathological manifestations of a 24-year-old woman who was initially diagnosed with rheumatoid arthritis. She presented to our institute with respiratory compromise and was found to have XD affecting skin, mucosal membranes, joints, and bone marrow. The patient received six cycles of cladribine for 6 months, during which she showed a remarkable response in relation to the respiratory lesions. Her hemoglobin also normalized and inflammatory markers gradually decreased to reach normal values. However, her skin lesions did not respond to treatment but no new lesions appeared. With our experience with cladribine, we believe that it could be a promising treatment option for XD. However, more work has to be conducted to determine the efficacy and safety in the long term. Key message: Given the rarity of this understudied entity, the natural history and the ultimate treatment remain unclear. We highlight the natural history and clinical course of xanthoma disseminatum (XD) in this article. We also describe our experience with cladribine in treating XD. We believe that similar experiences should be compiled to better understand this pathology and the effective therapy options for these patients.
format	article
author	Heba Al-Tarcheh Shahed Tish Salloum Salloum Ahed Haj Ibrahim
author_facet	Heba Al-Tarcheh Shahed Tish Salloum Salloum Ahed Haj Ibrahim
author_sort	Heba Al-Tarcheh
title	Xanthoma disseminatum with extensive respiratory involvement effectively treated with cladribine: a case report
title_short	Xanthoma disseminatum with extensive respiratory involvement effectively treated with cladribine: a case report
title_full	Xanthoma disseminatum with extensive respiratory involvement effectively treated with cladribine: a case report
title_fullStr	Xanthoma disseminatum with extensive respiratory involvement effectively treated with cladribine: a case report
title_full_unstemmed	Xanthoma disseminatum with extensive respiratory involvement effectively treated with cladribine: a case report
title_sort	xanthoma disseminatum with extensive respiratory involvement effectively treated with cladribine: a case report
publisher	Thieme Medical and Scientific Publishers Pvt. Ltd.
publishDate	2020
url	https://doaj.org/article/7d7f6313f38240c98387ccb037dab948
work_keys_str_mv	AT hebaaltarcheh xanthomadisseminatumwithextensiverespiratoryinvolvementeffectivelytreatedwithcladribineacasereport AT shahedtish xanthomadisseminatumwithextensiverespiratoryinvolvementeffectivelytreatedwithcladribineacasereport AT salloumsalloum xanthomadisseminatumwithextensiverespiratoryinvolvementeffectivelytreatedwithcladribineacasereport AT ahedhajibrahim xanthomadisseminatumwithextensiverespiratoryinvolvementeffectivelytreatedwithcladribineacasereport
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Xanthoma disseminatum with extensive respiratory involvement effectively treated with cladribine: a case report

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