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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Format: | Article |
Language: | English |
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Wolters Kluwer Medknow Publications
2008-01-01
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Series: | Indian Journal of Ophthalmology |
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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. |
first_indexed | 2024-12-22T14:23:01Z |
format | Article |
id | doaj.art-cdbec1d856ec43ca85933dfc53d16199 |
institution | Directory Open Access Journal |
issn | 0301-4738 1998-3689 |
language | English |
last_indexed | 2024-12-22T14:23:01Z |
publishDate | 2008-01-01 |
publisher | Wolters Kluwer Medknow Publications |
record_format | Article |
series | Indian Journal of Ophthalmology |
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 |
work_keys_str_mv | AT gulwanihanni isolatedlacrimalglandinvolvementinrosaidorfmandestombesdisease AT bhallasunita isolatedlacrimalglandinvolvementinrosaidorfmandestombesdisease AT srivastavaamit isolatedlacrimalglandinvolvementinrosaidorfmandestombesdisease |