IgG4-Related Disease and Malignancy

Yamamoto, Motohisa; Takahashi, Hiroki; Shinomura, Yasuhisa

doi:10.2169/internalmedicine.51.6782

Cited by 11 publications

(11 citation statements)

References 15 publications

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“…16 Malignancies have been documented at the time of diagnosis or during the course of disease. 16,21 However, none of our patients had malignancy at any point of time up to last follow-up (2-24 months). However, overall outcome of our patients was not so good.…”

Section: Discussionmentioning

confidence: 67%

“…Disease can relapse in about 20% of cases and minor adverse affects (worsening of diabetes, avascular necrosis, infections) not warranting any change in treatment or loss of patients have been documented in a follow‐up study . Malignancies have been documented at the time of diagnosis or during the course of disease . However, none of our patients had malignancy at any point of time up to last follow‐up (2–24 months).…”

Section: Discussionmentioning

confidence: 73%

“…IgG4‐RRD is part of spectrum of IgG4‐related systemic fibro‐inflammatory autoimmune diseases . Diagnostic criteria for IgG4TIN requires presence of clinical renal disease with histopathology showing TIN rich in plasma cells, that stain strongly for IgG4 (> 10 cells/HPF) with either serologic confirmation of elevated IgG4 or hypergammaglobunemia, radiology or histopathology confirmation of IgG4‐related disease in other organs.…”

Section: Discussionmentioning

confidence: 99%

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IgG4‐related tubulointerstitial nephritis: A prospective analysis

et al. 2015

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Aims: Immunoglobulin-G4 (IgG4)-related tubulo-interstitial nephritis (IgG4TIN) could be the first presentation of IgG4-related systemic disease. Most of the data is from the West or Japan and retrospective, with good patient outcome.Methods: This study was carried out from April 2011 to July 2013. We report a prospective follow-up of 11 patients who presented with renal dysfunction and had histological diagnosis of IgG4TIN followed for a minimum period of 1 year or until end-stage renal disease.Results: IgG4TIN constituted 0.28% of total renal biopsies and 6.5% of all tubulointerstitial nephritis. Patient ages ranged between 21 and 71 years with a male predominance. All the patients had renal dysfunction at presentation with a mean serum creatinine of 5.12 mg/dL. Proteinuria was subnephrotic except when there was coexisting membranous glomerulonephritis (36.4%). The mean 24-h urine protien excretion was 1.8 g. Serum IgG4 levels were elevated in 10 (90.9%) patients. Ten (90.9%) patients had renomegaly and one (9.1%) had focal renal mass. Extra-renal manifestations were present in seven (63.6%). Renal histology showed pattern A in five (45.5%), pattern B in four (36.3%) and pattern C in two (18.1%) patients. All but one patient (90.9%) received immunosuppressive therapy. Four (36.3%) achieved complete remission and three (27.2%) progressed to end stage renal disease. Two patients died due to infections while on steroid therapy. One patient with a mass had end stage renal disease for 12 months and did not improve with steroid therapy, and one (pattern C) had progressive chronic kidney disease on follow-up.Conclusion: IgG4TIN in an Indian cohort most often presents with rapidly progressive renal failure and less often has extra-renal organ involvement. On follow-up, patients can experience a more aggressive course with progression to end stage renal disease.

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Section: Discussionmentioning

confidence: 67%

Section: Discussionmentioning

confidence: 73%

Section: Discussionmentioning

confidence: 99%

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IgG4‐related tubulointerstitial nephritis: A prospective analysis

et al. 2015

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“…A link between ocular adnexal IgG4-related disease and EMZL has been proposed [ 17 , 18 ]. EMZL can be caused by chronic infection or inflammation [ 19 – 21 ].…”

Section: Discussionmentioning

confidence: 99%

Clinicopathological Analysis of Ocular Adnexal Extranodal Marginal Zone B-Cell Lymphoma with IgG4-Positive Cells

et al. 2015

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This study aims to analyze clinical and pathological characteristics of ocular adnexal extranodal marginal zone B-cell lymphoma (EMZL) accompanying IgG4-positive cells. Fifty patients with a diagnosis of primary non-conjunctival ocular adnexal EMZL were enrolled in this study. The number of IgG4-positive cells and the ratio of IgG/IgG4 were evaluated by immunohistochemistry in the biopsy specimens. The patients were divided into two groups based on the absolute number and the ratio of IgG4-positive cells (IgG4-posivite vs IgG4-negative groups). The demographic data, clinical staging at diagnosis, histopathological characteristics, and response to initial treatment were comparatively analyzed between the 2 groups. Five (10%) of 50 patients were defined as IgG4-positive group, and all the cases showed characteristic histological features such as extensive plasma cell infiltration and dense fibrosis. Most of these patients (4 of 5 patients) had lymphoma of the lacrimal gland. The patients from the IgG4-positive group showed a lower response rate to initial treatment (87.5 vs 33%, p = 0.03) than IgG4-negative group with a median follow-up period of 38 months. A part of the ocular adnexal EMZLs were accompanied with IgG4-positive cells. Significantly, most IgG4-positive ocular adnexal EMZLs occurred in the lacrimal gland, and can be related with a more frequent treatment failure.

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“…[ 6 ] In this context, PET (positron emission tomography) could be useful for both systemic evaluation in IgG4-RD and cancer detection. [ 26 , 27 ] PET was not performed in our patient. However, given that the prevalence of certain cancers is high in the elderly population (in particular colon cancer), a chance association between IgG4-RD and cancer remains possible and more epidemiological studies are needed to study the relationship between the two.…”

Section: Discussionmentioning

confidence: 95%

Colon cancer and IgG4-related disease with orbital inflammation and bilateral optic perineuritis

Lemaître

Esquerda²,

Guardiola

et al. 2018

Medicine

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Rationale:Immunoglobulin G4-related disease (IgG4-RD) is an inflammatory condition of unknown cause. Cancer might be related to the development of certain IgG4-RD but to date, little literature documents it.Patient concerns:A 78-year old man presented with unilateral proptosis responsive to steroids, initially attributed to nonspecific orbital inflammation.Diagnosis:Right hemicolectomy was performed because of a suspicious lesion which turned out to be tubulovillous adenoma on histological analysis. Eight months after the surgery, a mass infiltrating the mesentery was found and biopsy revealed IgG4-RD.Interventions:Both the orbital inflammation and abdominal mass infiltrating the mesentery were responsive to steroids and rituximab administered to treat IgG4-RD.Outcomes:In the course of IgG4-RD, the patient developed bilateral optic perineuritis, causing bilateral visual loss. Colon cancer with synchronous multiple liver metastases was found 1 year after rituximab treatment.Lessons:This case raises the possibility of IgG4-RD being a paraneoplastic syndrome in some patients. Cancer screening should probably be performed in some elderly patients diagnosed with IgG4-RD.

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IgG4-Related Disease and Malignancy

Cited by 11 publications

References 15 publications

IgG4‐related tubulointerstitial nephritis: A prospective analysis

IgG4‐related tubulointerstitial nephritis: A prospective analysis

Clinicopathological Analysis of Ocular Adnexal Extranodal Marginal Zone B-Cell Lymphoma with IgG4-Positive Cells

Colon cancer and IgG4-related disease with orbital inflammation and bilateral optic perineuritis

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