Incidence of Malignancies in Patients with IgG4-related Disease

Hirano, Keisuke; Tada, Minoru; Sasahira, Naoki; Isayama, Hiroyuki; Mizuno, Suguru; Takagi, Kaoru; Watanabe, Takeo; Saito, Tomotaka; Kawahata, Shuhei; Uchino, Rie; Hamada, Tsuyoshi; Miyabayashi, Koji; Mohri, Dai; Sasaki, Takashi; Kogure, Hirofumi; Yamamoto, Natsuyo; Yoshida, Haruhiko; Ito, Yukiko; Akiyama, Dai; Toda, Nobuo; Arizumi, Toshihiko; Yagioka, Hiroshi; Takahara, Naminatsu; Matsubara, Saburo; Yashima, Yoko; Koike, Kazuhiko

doi:10.2169/internalmedicine.53.1342

Cited by 158 publications

(144 citation statements)

References 22 publications

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“…On the other hand, we previously concluded that IgG4-RD was not associated with an increased incidence of total malignancy [14]. In our experience of 126 patients with IgG4-RD, there were 23 malignancies among 21 patients, which might not be a small figure.…”

Section: Aip Igg4-rd and Total Malignanciesmentioning

confidence: 50%

“…As for IPMN, pancreatic cancer was discovered in 2.6 % (17/642) during an average follow-up of 4.8 years in one study [13]. If 100 patients with AIP are necessary to identify one case of pancreatic cancer and the SIR is 3.6, as per our previous report [14], a sample of approximately 450 patients is necessary to demonstrate statistical significance [15]. It is possible that the SIR is much \ 3.6 [16].…”

Section: Aip and Pancreatobiliary Cancermentioning

confidence: 70%

“…Concerning AIP and total malignancy, it is also difficult to draw any specific conclusions given our current understanding. In light of the results of two reports [14,16], however, it seems unlikely that AIP is responsible for the increased risk of total malignancy. Shiokawa et al…”

Section: Discussionmentioning

confidence: 99%

“…If there were prodromal symptoms before the final diagnosis of IgG4-RD, the timing of the appearance of the symptoms was regarded as the onset time in our study [14]. However, in patients without any symptoms, the onset time was regarded as the time of diagnosis.…”

Section: Institutional Biasmentioning

confidence: 99%

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Association between autoimmune pancreatitis and malignancy

Hirano

Isayama

Tada

et al. 2014

Clin J Gastroenterol

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Chronic inflammatory diseases are often associated with malignancy, and this phenomenon occurs even in the field of gastroenterology. For example, chronic pancreatitis has been reported to be highly associated with the occurrence of pancreatic cancer. However, the association between autoimmune pancreatitis (AIP) and pancreatic cancer is still an unsolved problem. As AIP is associated with immunoglobulin G4-related disease, which causes a variety of inflammatory and fibrotic lesions, the association between AIP and total malignancies is also a matter of interest. Here, we discuss the association between AIP and malignancy. As for the association between AIP and pancreatic cancer, there is no conclusive evidence, although several reports have suggested that the relative risk of pancreatic cancer is high in AIP. Concerning the association between AIP and total malignancies, there are two reports from Japan. Neither report supported the idea that chronic inflammation in AIP caused the increased risk of total malignancies. However, one report proposed the possibility that AIP might develop as a paraneoplastic syndrome in some patients because many cancers occurred within 1 year of AIP diagnosis, and thereby concluded that patients with AIP were at high risk of having various cancers. The paraneoplastic syndrome hypothesis should be tested in future studies.

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Section: Aip Igg4-rd and Total Malignanciesmentioning

confidence: 50%

Section: Aip and Pancreatobiliary Cancermentioning

confidence: 70%

Section: Discussionmentioning

confidence: 99%

Section: Institutional Biasmentioning

confidence: 99%

See 2 more Smart Citations

Association between autoimmune pancreatitis and malignancy

Hirano

Isayama

Tada

et al. 2014

Clin J Gastroenterol

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“…Patients with IgG4-RD were reported to be at risk of malignant disease [2]. However some researchers found that the incidence of total malignancies in IgG4-RD patients was similar to that in the general population [9]. One thing is sure that IgG4-RD should be differentiated from malignant diseases.…”

Section: Discussionmentioning

confidence: 99%

The Expression of IgG and IgG4 in Lymphoma

Li¹,

Zhang²

2014

Intern Med

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Objectives: Although IgG4-related disease has been gradually recognized, its relationship with malignant diseases, especially lymphoma has been an eternal topic. Therefore we set out to explore the expression of IgG4 positive cells in lymphoma.Methods: Surgical excision specimens with definite diagnosis of lymphoma from January to December, 2013 were collected. Hematoxylin-eosin staining and immunohistochemical staining of IgG and IgG4 were then evaluated on dense lymphoplasmacytic infiltration, storiform fibrosis and obliterative phlebitis. For the quantification of IgG and IgG4 positive cells, the areas with the highest density of positive cells were evaluated. Three high-powered fields (hpf) in each section were analyzed, and the average number of positive cells per hpf was calculated.Results: 16 patients with lymphoma were included in our study. There were 9 males and 7 females with an average age of 51 years old. 3 cases were diagnosed as Hodgkin lymphoma while 13 were non-Hodgkin lymphoma (diffuse large B cell lymphoma 8 cases, small B cell lymphoma 2 cases, mucosa associated lymphoid tissue marginal zone B cell lymphoma (MALToma) 1 case, follicular lymphoma, peripheral T-cell lymphoma and hepatosplenic T-cell lymphoma 1 case. Dense lymphocytic infiltration accompanied by atypical lymphocytes was observed in all the specimens. Proliferation of fibrous tissue was only seen in one specimen. IgG positive cells were detectable in 14 cases with the highest cell count from 20-350/hpf. 2 Hodgkin lymphoma cases showed of IgG4 positivity with the highest cell counts were 11 and 12/hpf respectively. Conclusion:IgG4 positive cells, fibrosis and obliterative phlebitis barely appeared in lymphoma. In combination with specific tumor signature molecules, it may not be difficult to distinguish lymphoma from IgG4-related disease.

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Association of Rituximab Use With Adverse Events in Children, Adolescents, and Young Adults

et al. 2021

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IMPORTANCE Rituximab is among the most frequently used immunotherapies in pediatrics. Few studies have reported long-term adverse events associated with its use for children. OBJECTIVE To describe the use of rituximab and to assess whether its use is associated with shortor long-term adverse events, infections, or time to immune reconstitution in a diverse group of young people. DESIGN, SETTING, AND PARTICIPANTS This retrospective cohort study included 468 patients aged younger than 21 years who received rituximab for diverse indications between October 1, 2010, and December 31, 2017, at Texas Children's Hospital, a large pediatric referral hospital. Patterns of adverse events, infections, and immune recovery are described. Data analyses were conducted from December 2019 to June 2020. EXPOSURE One or more doses of rituximab. MAIN OUTCOMES AND MEASURES Adverse drug events (eg, anaphylaxis), incidence of mild and severe infections, and time to recovery of B lymphocyte subset counts and immunoglobulin levels. Survival models and logistic regression analyses and were used to identify associated risk factors of infectious and noninfectious adverse drug events. RESULTS We identified 468 patients receiving at least 1 dose of rituximab. The total follow-up time was 11 713 person-months. Of the 468 patients, 293 (62.6%) were female, the median (interquartile range) age at receipt of dose was 14.3 (9.9-16.8) years, and 209 (44.7%) were self-reported White Hispanic. Adverse events associated with rituximab infusion occurred in 72 patients (15.4%), and anaphylaxis occurred in 17 patients (3.6%). Long-term adverse events, such as prolonged neutropenia and leukoencephalopathy, were absent. Infections occurred in 224 patients (47.9%); 84 patients (17.9%) had severe infections, and 3 patients (0.6%) had lethal infections. Concurrent use of intravenous chemotherapy, treatment of systemic lupus erythematosus, neutropenia, and use of intravenous immunoglobulin were associated with increased risk of infection. Among 135 patients (28.8%) followed up to B cell count recovery, CD19 + or CD20 + cell numbers normalized in a median of 9.0 months (interquartile range, 5.9-14.4 months) following rituximab use; 48 of 95 patients (51%) evaluated beyond a year had low-forage B cell counts. Recovery of CD27 + memory B cell number occurred in a median of 15.7 months (interquartile range, 6.0-22.7 months). Among patients with normal baseline values, low immunoglobulin G (IgG) levels developed in 67 of 289 patients (23.2%) and low IgM levels in 118 of 255 patients (40.8%); of these patients evaluated beyond 12 months from rituximab, 16 of 117 (13.7%) had persistently low IgG and 37 (33.9%) of 109 had persistently low IgM.

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Incidence of Malignancies in Patients with IgG4-related Disease

Cited by 158 publications

References 22 publications

Association between autoimmune pancreatitis and malignancy

Association between autoimmune pancreatitis and malignancy

The Expression of IgG and IgG4 in Lymphoma

Association of Rituximab Use With Adverse Events in Children, Adolescents, and Young Adults

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