Mucinous Cystic Neoplasm of the Pancreas With Increased IgG4+ Plasma Cells and Histopathologic Features of Autoimmune Pancreatitis/IgG4—Related Disease

Yakirevich, Evgeny; Henriksen, Kammi J.; Miner, Thomas J.; Resnick, Murray B.

doi:10.1097/mpa.0000000000000296

Cited by 9 publications

(5 citation statements)

References 10 publications

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“…With the normal pancreatic ductal system observed on radiologic imaging, these features can be interpreted as indicating a localized form of AIP1. Similar features were reported in the previous case [12]. Comparisons of clinicopathologic characteristics of MCN in association with AIP1 with previously reported cases and the present case are summarized in Table 1.…”

Section: Discussionsupporting

confidence: 87%

“…There have been several case reports about AIP1 accompanied by other pancreatic lesions, such as a pseudocyst or IPMN [8-11]. However, MCN with AIP1 is extremely rare and only two cases have been reported in the English-language literature [12,13]. Here, we report a third case, a patient with both low-grade MCN and AIP1.…”

mentioning

confidence: 78%

“…All the reported cases exhibited the histologic features of AIP1; in addition, some cases showed increased serum IgG4 levels [8,9,11], whereas another did not [10]. There has been only one English-language report of a case of MCN accompanied by the histologic features of AIP1, and that case had an elevated serum IgG4 level [12].…”

Section: Discussionmentioning

confidence: 99%

“…The most plausible explanation is the co-existence of two lesions, the AIP1 and the MCN [13]. Other possibilities include a secondary inflammatory response to a pre-existing MCN due to the use of fine needle aspiration biopsy or cytology of the MCN for histologic confirmation, or a secondary immune reaction of the periphery of the cyst wall of the pancreas to inspissated extracellular mucin from the MCN [12].…”

Section: Discussionmentioning

confidence: 99%

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Coexisting Mucinous Cystic Neoplasm of the Pancreas and Type 1 Autoimmune Pancreatitis

Kim

Song

Kim

et al. 2019

J Pathol Transl Med

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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.

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Section: Discussionsupporting

confidence: 87%

mentioning

confidence: 78%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Coexisting Mucinous Cystic Neoplasm of the Pancreas and Type 1 Autoimmune Pancreatitis

Kim

Song

Kim

et al. 2019

J Pathol Transl Med

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“…IgG4-related disease is a disease associated with serum increased IgG4, which is involved in chronic and autoimmune disease in multiple organs and/or tissues (Grados et al 2015 ), including many kinds of diseases (Furukawa et al 2015 ; Yakirevich et al 2015 ; Nishikawa et al 2014 ; Kragstrup et al 2013 ; Ahn et al 2014 ; Huggett et al 2014 ; Pusztaszeri et al 2012 ; Hori et al 2010 ), for example, inflammatory pseudotumor (Choi et al 2015 ), Mikulicz`s disease (Takano et al 2014 ), and autoimmune pancreatitis (AIP) (Yakirevich et al 2015 ). AIP often expresses acute or chronic pancreatitis and imaging scanning shows characteristic sausage-type swelling of the pancreas (Hara et al 2014 ; Brito-Zerón et al 2014 ; Eu Jin et al 2010 ).…”

Section: Discussionmentioning

confidence: 99%

IgG4-related disease: a case report with duration of more than 16 years and review of literature

Peng

Xie

et al. 2016

SpringerPlus

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IntroductionIgG4-related disease was first reported in 2001 and was officially named in 2010. It is now considered as a systemic disease that might affect any organ system. The characteristic pathological changes of IgG4-related disease are extensive infiltration of IgG4-positive plasma cells. IgG4-related disease is a kind of benign lesions, but there has not been well-defined standard treatment so far. Patients usually respond well to corticosteroids. The prognosis of IgG4-related disease is perhaps good as long as early detection and treatment.Case descriptionWe report one case of IgG4-related disease with a 16-years anamnesis with multi-pseudotumor masses. He was diagnosed with chronic kidney disease with wide interstitial renal fibrosis. And he received glucocorticoids therapy. After 2 month therapy, the serum creatinine, erythrocyte sedimentation rate, and serum IgG4 decreased significantly. The discussion includes presentation, clinical course, diagnosis, and prognosis of IgG4-related disease. The case and discussion highlight the importance of diagnosis and the good prognosis of IgG4-related diseases.Discussion and evaluationOur case highlights the importance of diagnosis and the good prognosis of IgG4-related diseases. IgG4-related disease is a systemic fibro-inflammatory immune-mediated disorder and now recognized in almost every major organs. Characteristics of the disease is multiple lymph nodes and the response to glucocorticoids therapy is well. In such case, he had a history of 16 years with multi-pseudotumor masses and misdiagnosed for 16 years, if the doctors were not awareness of higher serum immunoglobulin G4 (IgG4) than normal, the correct diagnosis may be missed or delayed. Consequently, appropriate treatment for IgG4-related disease would also be delayed or not provided and likely result in increased morbidity and mortality.ConclusionsIgG4-related disease is a systemic fibro-inflammatory immune-mediated disorder and progresses slowly. In the present patient the course of IgG4-related disease appears to be benign. The prognosis of IgG4-related disease depend on early diagnosis and treatment.

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Histological features of autoimmune pancreatitis and IgG4-related sclerosing cholangitis with a correlation with imaging findings

Notohara

2021

J Med Ultrasonics

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Mucinous Cystic Neoplasm of the Pancreas With Increased IgG4+ Plasma Cells and Histopathologic Features of Autoimmune Pancreatitis/IgG4—Related Disease

Cited by 9 publications

References 10 publications

Coexisting Mucinous Cystic Neoplasm of the Pancreas and Type 1 Autoimmune Pancreatitis

Coexisting Mucinous Cystic Neoplasm of the Pancreas and Type 1 Autoimmune Pancreatitis

IgG4-related disease: a case report with duration of more than 16 years and review of literature

Histological features of autoimmune pancreatitis and IgG4-related sclerosing cholangitis with a correlation with imaging findings

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