Isolated lacrimal gland involvement in Rosai-Dorfman-Destombes disease

Rosai-Dorfman-Destombes (sinus histiocytosis with massive lymphadenopathy) disease is an uncommon disease characterized by benign proliferation of histiocytes, with painless lymph node enlargement and frequent extranodal disease. Orbital involvement occurs in 9-11% of cases. However, isol...

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Main Authors: Gulwani Hanni, Bhalla Sunita, Srivastava Amit
Format: Article
Language:English
Published: Wolters Kluwer Medknow Publications 2008-01-01
Series:Indian Journal of Ophthalmology
Subjects:
Online Access:http://www.ijo.in/article.asp?issn=0301-4738;year=2008;volume=56;issue=6;spage=507;epage=509;aulast=Gulwani
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author Gulwani Hanni
Bhalla Sunita
Srivastava Amit
author_facet Gulwani Hanni
Bhalla Sunita
Srivastava Amit
author_sort Gulwani Hanni
collection DOAJ
description Rosai-Dorfman-Destombes (sinus histiocytosis with massive lymphadenopathy) disease is an uncommon disease characterized by benign proliferation of histiocytes, with painless lymph node enlargement and frequent extranodal disease. Orbital involvement occurs in 9-11% of cases. However, isolated Rosai-Dorfman-Destombes disease of the lacrimal gland without any systemic involvement is very rare with only three case reports. We describe here one such young male patient with unilateral lacrimal gland swelling. Excision biopsy revealed almost complete replacement of the lacrimal gland by lymphocytes, plasma cells and large pale histiocytes. The latter exhibited emperipolesis and stained positive for S-100 and CD68 on immunohistochemistry. Patient is well and has no other manifestation or recurrence of the disease during a follow-up of 24 months.
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spelling doaj.art-cdbec1d856ec43ca85933dfc53d161992022-12-21T18:22:56ZengWolters Kluwer Medknow PublicationsIndian Journal of Ophthalmology0301-47381998-36892008-01-01566507509Isolated lacrimal gland involvement in Rosai-Dorfman-Destombes diseaseGulwani HanniBhalla SunitaSrivastava AmitRosai-Dorfman-Destombes (sinus histiocytosis with massive lymphadenopathy) disease is an uncommon disease characterized by benign proliferation of histiocytes, with painless lymph node enlargement and frequent extranodal disease. Orbital involvement occurs in 9-11% of cases. However, isolated Rosai-Dorfman-Destombes disease of the lacrimal gland without any systemic involvement is very rare with only three case reports. We describe here one such young male patient with unilateral lacrimal gland swelling. Excision biopsy revealed almost complete replacement of the lacrimal gland by lymphocytes, plasma cells and large pale histiocytes. The latter exhibited emperipolesis and stained positive for S-100 and CD68 on immunohistochemistry. Patient is well and has no other manifestation or recurrence of the disease during a follow-up of 24 months.http://www.ijo.in/article.asp?issn=0301-4738;year=2008;volume=56;issue=6;spage=507;epage=509;aulast=GulwaniLacrimal glandRosai-Dorfman-Destombes diseasesinus histiocytosis with massive lymphadenopathy
spellingShingle Gulwani Hanni
Bhalla Sunita
Srivastava Amit
Isolated lacrimal gland involvement in Rosai-Dorfman-Destombes disease
Indian Journal of Ophthalmology
Lacrimal gland
Rosai-Dorfman-Destombes disease
sinus histiocytosis with massive lymphadenopathy
title Isolated lacrimal gland involvement in Rosai-Dorfman-Destombes disease
title_full Isolated lacrimal gland involvement in Rosai-Dorfman-Destombes disease
title_fullStr Isolated lacrimal gland involvement in Rosai-Dorfman-Destombes disease
title_full_unstemmed Isolated lacrimal gland involvement in Rosai-Dorfman-Destombes disease
title_short Isolated lacrimal gland involvement in Rosai-Dorfman-Destombes disease
title_sort isolated lacrimal gland involvement in rosai dorfman destombes disease
topic Lacrimal gland
Rosai-Dorfman-Destombes disease
sinus histiocytosis with massive lymphadenopathy
url http://www.ijo.in/article.asp?issn=0301-4738;year=2008;volume=56;issue=6;spage=507;epage=509;aulast=Gulwani
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AT srivastavaamit isolatedlacrimalglandinvolvementinrosaidorfmandestombesdisease