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Case Reports
Rosai-Dorfman Disease: Report of a Case Associated with IgG4-Related Sclerotic Lesions
Bong-Hee Park, Da Hye Son, Myung-Hwan Kim, Tae Sun Shim, Hee Jin Lee, Jooryung Huh
Korean J Pathol. 2012;46(6):583-586.   Published online December 26, 2012
DOI: https://doi.org/10.4132/KoreanJPathol.2012.46.6.583
  • 6,869 View
  • 51 Download
  • 5 Crossref
AbstractAbstract PDF

We describe a rare case of sinus histiocytosis with massive lymphadenopathy (Rosai-Dorfman disease) associated with a six-year history of autoimmune pancreatitis, which was controlled by steroid treatment. The patient presented with multiple, cervical and thoracic lymphadenopathy and abnormal, nodular opacities in the lung. Histologically, Rosai-Dorfman disease with numerous IgG4-positive cells was identified in a subcutaneous lymph node in the patient's left forearm. The patient recovered uneventfully with steroid treatment.

Citations

Citations to this article as recorded by  
  • Lung Involvement in Destombes-Rosai-Dorfman Disease
    Quentin Moyon, Samia Boussouar, Philippe Maksud, Jean-François Emile, Frédéric Charlotte, Nathalie Aladjidi, Grégoire Prévot, Jean Donadieu, Zahir Amoura, Philippe Grenier, Julien Haroche, Fleur Cohen Aubart
    Chest.2020; 157(2): 323.     CrossRef
  • Rosai-Dorfman Disease: Rare Pulmonary Involvement Mimicking Pulmonary Langerhans Cell Histiocytosis and Review of the Literature
    Rashid AL Umairi, Danielle Blunt, Wedad Hana, Matthew Cheung, Anastasia Oikonomou
    Case Reports in Radiology.2018; 2018: 1.     CrossRef
  • IgG4‐related skin disease may have distinct systemic manifestations: a systematic review
    Adam E. Bennett, Neil A. Fenske, Paul Rodriguez‐Waitkus, Jane L. Messina
    International Journal of Dermatology.2016; 55(11): 1184.     CrossRef
  • Nosology and Pathology of Langerhans Cell Histiocytosis
    Jennifer Picarsic, Ronald Jaffe
    Hematology/Oncology Clinics of North America.2015; 29(5): 799.     CrossRef
  • A subset of Rosai–Dorfman disease cases show increased IgG4‐positive plasma cells: another red herring or a true association with IgG4‐related disease?
    Madhu P Menon, Moses O Evbuomwan, Juan Rosai, Elaine S Jaffe, Stefania Pittaluga
    Histopathology.2014; 64(3): 455.     CrossRef
Rosai-Dorfman Disease of the Nose and Salivary Gland: A case report.
Mee Sook Roh, Jin Sook Jeong, Sook Hee Hong
Korean J Pathol. 1999;33(12):1203-1206.
  • 1,607 View
  • 16 Download
AbstractAbstract PDF
Rosai-Dorfman disease (RDD) is a rare type of benign histiocytosis characterized histologically by intracellular engulfment of lymphocytes. Extranodal RDD may occur as a part of generalized process involving lymph nodes or may involve extranodal sites independent of the lymph node status. We have experienced a case of extranodal Rosai-Dorfman disease of the nose as an initial lesion prior to nodal involvement. The patient was a 20-year-old woman who complained of nasal obstruction for 4 years, remotely, and left submandibular mass for 3 months, recently. Histologically, the lesion taken from nasal cavity, submandibular gland and left upper jugular lymph node all showed an heavy infiltrate consisted of plasma cells, lymphocytes and sheets of macrophages with abundant pale cytoplasm, which replaced organ architecture. The associated focal fibrosis made it difficult to differentiate from inflammatory pseudotumor. Some macrophages demonstrated phagocytosis of lymphocytes, plasma cells and occasionally neutrophils. The macrophages were strongly positive for S-100 protein.

J Pathol Transl Med : Journal of Pathology and Translational Medicine