Journal of Pathology and Translational Medicine

Case Study

Coexisting Mucinous Cystic Neoplasm of the Pancreas and Type 1 Autoimmune Pancreatitis: Mee-Jeong Kim, Tae Jun Song, Hyoung Jung Kim, Song-Cheol Kim, Myung-Hwan Kim, Seung-Mo Hong; J Pathol Transl Med. 2019;53(2):125-128. Published online November 14, 2018; DOI: https://doi.org/10.4132/jptm.2018.10.25

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Type 1 autoimmune pancreatitis (AIP1) is an IgG4-related systemic disease that mimics tumors. We report a rare case of AIP1 accompanied by mucinous cystic neoplasm (MCN). A pancreatic lesion was incidentally detected in a woman in her 60s. After 6 years of follow-up, the lesion abruptly increased in size. Computed tomography showed a 3.5 cm unilocular cyst in the tail of the pancreas and distal pancreatectomy was performed. On microscopic examination, the cyst was lined by mucinous and non-mucinous epithelial cells with mild cytologic atypia. The surrounding stroma comprised ovarian-type spindle cells with progesterone receptor positivity. The pericystic pancreas exhibited multifocal lymphoid follicles, lymphoplasmacytic infiltrations, obliterative phlebitis, and storiform fibrosis. IgG4-positive plasma cell infiltration (215 cells high-power field) and the IgG4/IgG ratio (57%) were increased. Cases of MCN coexisting with AIP1 are extremely rare; only two such cases have been reported in the English-language literature. This third case featured low-grade MCN with AIP1.

Citations

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Case Report

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

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Abstract 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

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