The front line of research into immunoglobin G4-related disease - Do autoantibodies cause immunoglobin G4-related disease?

Umehara, Hisanori; Okazaki, Kazuichi; Kawano, Mitsuhiro; Tanaka, Yoshiya

doi:10.1080/14397595.2018.1558519

Cited by 25 publications

(16 citation statements)

References 39 publications

(54 reference statements)

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“…These findings can also be supported by a recently reported co‐occurrence of IgG4‐RH and a craniopharyngioma 42 . Genetic predisposition and presence of autoantibodies have already been suggested as mechanisms responsible for the development of IgG4‐RD 43‐45 …”

Section: Discussionsupporting

confidence: 77%

Histopathological findings in the landscape of IgG4‐related pathology in patients with pituitary dysfunction: Review of six cases

Vasaitis

Wikström

Ahlström

et al. 2021

J Neuroendocrinology

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IgG4-related hypophysitis (IgG4-RH) is increasingly being reported as an isolated entity or, less frequently, as a manifestation of a multiorgan IgG4-related disease (IgG4-RD), in which typical histopathology is a cornerstone for the diagnosis. We aimed to describe the histopathological changes in the surgical specimens from patients with clinical signs of pituitary disease that fulfilled the current diagnostic criteria for IgG4-RH. Histopathological features were correlated with clinical and radiological findings. Of 19 patients with pituitary dysfunction and inflammatory changes in the surgical pituitary specimen operated on during 2011-2019, we identified five patients with typical IgG4-related pathology (lymphoplasmacytic infiltration with more than 10 IgG4-positive plasma cells per one high power microscopic field, representing at least 40% of all plasma cells and at least focal storiform fibrosis). One patient with diabetes insipidus and pachymeningitis with IgG4-related changes in a biopsy from the dura was also included. Additional histopathological changes that typically are not part of the IgG4-RH were observed: Rathke's cleft cyst in four and granulomatous changes in two patients. One patient had an elevated serum IgG4 level and systemic manifestations that could be associated with the systemic IgG4-RD. Our findings indicate that pure IgG4-RH is uncommon. All patients with pituitary dysfunction, beyond typical IgG4-related pathology, had other pathological findings that could trigger the secondary IgG4-response. Both primary pathology and secondary IgG4-related features should be reported in patients with pituitary dysfunction because their co-occurrence may cause atypical clinical and imaging features, and unexpected response to surgical and pharmacological treatment. The current criteria for the diagnosis of IgG4-RH can lead to overdiagnosis of IgG4-RH if additional pathological changes are not taken into consideration. The classification criteria of IgG4-RD proposed by the American College of Rheumatology/European League Against Rheumatism could help classify patients more properly as IgG4-RH if applied to the pituitary gland.

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Section: Discussionsupporting

confidence: 77%

Histopathological findings in the landscape of IgG4‐related pathology in patients with pituitary dysfunction: Review of six cases

Vasaitis

Wikström

Ahlström

et al. 2021

J Neuroendocrinology

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“…The PD-1/PD-L1 pathway also contributes to the interaction between T follicular helper cells and plasmablasts [9]. One possible mechanism is that the inhibition of PD-1/PD-L1 pathway enhances T follicular helper cells to promote the growth, differentiation, and class switching of B cells to IgG4 [11,12]. Another possibility is that there is an association between pulmonary adenocarcinoma and IgG4-related disease.…”

Section: Discussionmentioning

confidence: 99%

IgG4-related pleural disease in a patient with pulmonary adenocarcinoma under durvalumab treatment: a case report

et al. 2020

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Background: Immune checkpoint inhibitors (ICIs) are the standard treatment for non-small cell lung cancer. The unique adverse events that can arise after treatment with ICIs are known as immune-related adverse events (irAE). As the number of cases under treatment with ICIs increases, new types of characteristics of irAE have emerged. This case report suggests that IgG4-related pleural disease could occur as an irAE. Case presentation: A 64-year-old man was diagnosed with pulmonary adenocarcinoma stage IIIB. Following concurrent chemoradiotherapy, durvalumab was administered every two weeks. The patient complained of dyspnea on effort 4 months after the initiation of durvalumab therapy. Chest CT scans showed mild bilateral pleural effusion 4 months after the initiation of durvalumab therapy, and the amount of pleural effusion increased further at 7 months. Durvalumab was thought to be a potential cause of pleural effusion and was withdrawn after 13 courses of administration over 7 months. The level of serum IgG4 was 2750 mg/dL. The levels of IgG4 of the pleural fluids were 2790 mg/dL on the right side and 2890 mg/dL on the left side at 7 months. Microscopic examination of the pleural biopsy revealed lymphoplasmacytic infiltration with storiform fibrosis. Immunohistochemical examinations showed that the number of IgG4-positive cells was > 20/high power field and the percentage of IgG4-positive to IgG-positive plasma cells was > 50%. Oral prednisolone at a dose of 30 mg/day was initiated, and remarkable clinical improvements were achieved. After 4 months of prednisolone therapy, the level of serum IgG4 decreased to 370 mg/dL and chest CT revealed the disappearance of bilateral pleural effusion. Conclusion: This was a case of IgG4-related pleural disease in a patient with pulmonary adenocarcinoma under durvalumab treatment. To our knowledge, this is the first case report of IgG4-related pleural disease as an irAE. It is important to consider the possibility of IgG4-related pleural disease in cases of pleural effusion during the treatment with ICIs.

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“…Numerous self-antigens have been reported as potential autoantigens responsible for the pathogenesis of IgG4-RD [31]. However, it is unclear whether IgG4 antibodies are pathogenic in IgG4-RD or merely represent a regulatory response to another primary autoimmune or allergic process [1,19,[31][32][33][34][35]. In an experimental animal model, J Investig Allergol Clin Immunol 2021; Vol.…”

Section: Etiology and Pathophysiology Of Igg4-related Diseasementioning

confidence: 99%

Immunoglobulin G4–Related Disease: What an Allergist Should Know

Carballo

González‐Quintela

Sopeña

et al. 2021

J Investig Allergol Clin Immunol

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The front line of research into immunoglobin G4-related disease - Do autoantibodies cause immunoglobin G4-related disease?

Cited by 25 publications

References 39 publications

Histopathological findings in the landscape of IgG4‐related pathology in patients with pituitary dysfunction: Review of six cases

Histopathological findings in the landscape of IgG4‐related pathology in patients with pituitary dysfunction: Review of six cases

IgG4-related pleural disease in a patient with pulmonary adenocarcinoma under durvalumab treatment: a case report

Immunoglobulin G4–Related Disease: What an Allergist Should Know

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