title: Isolated lacrimal gland involvement in Rosai-Dorfman-Destombes disease.
abstract: 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.
authors.affiliation: Department of Histopathology, Sir Ganga Ram Hospital, New Delhi, India. hannigulwani@yahoo.com
authors.forename: Hanni
authors.initials: H
authors.lastname: Gulwani
authors.forename: Sunita
authors.initials: S
authors.lastname: Bhalla
authors.forename: Amit
authors.initials: A
authors.lastname: Srivastava
mesh_terms: D000328:Adult; D015703:Antigens, CD / Q000378:metabolism; D015214:Antigens, Differentiation, Myelomonocytic / Q000378:metabolism; D006644:Histiocytes / Q000378:metabolism / Q000473:pathology; D015618:Histiocytosis, Sinus / Q000150:complications* / Q000175:diagnosis / Q000378:metabolism; D006801:Humans; D007124:Immunoenzyme Techniques; D007766:Lacrimal Apparatus Diseases / Q000175:diagnosis / Q000209:etiology* / Q000378:metabolism; D008214:Lymphocytes / Q000473:pathology; D008279:Magnetic Resonance Imaging; D008297:Male; D010950:Plasma Cells / Q000473:pathology; D009418:S100 Proteins / Q000378:metabolism; D000097382:CD68 Molecule
chemical_list: D015703:Antigens, CD; D015214:Antigens, Differentiation, Myelomonocytic; D009418:S100 Proteins; C067980:CD68 antigen, human; D000097382:CD68 Molecule