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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Bibliographic Details
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
Description
Summary: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.
ISSN:0301-4738
1998-3689