Sclerosing sialadenitis of the submandibular gland is rarely an immunoglobulin G4-related disease in the Finnish population

Peuraharju, Elin; Saarinen, Riitta; Aro, Katri; Mäkinen, Laura K.; Tarkkanen, Jussi; Mäkitie, Antti; Haglund, Caj; Hagström, Jaana; Atula, Timo

doi:10.1038/s41379-019-0395-5

Cited by 14 publications

(20 citation statements)

References 25 publications

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“…A possible explanation for this is that IgG4‐RD was first identified in Japan, Hamano et al, 2001 and there is a low prevalence and poor recognition of IgG4‐RS as a clinical entity in Western countries. Gunasekara et al, 2016; Peuraharju et al, 2020 This may imply a certain racial disparity in the incidence of IgG4‐RS, as indicated by Wallace et al Wallace et al, 2019 Third, a universally accepted organ‐specific diagnosis criterion for IgG4‐RD has not been established, and a histological analysis of the biopsy of each organ was not conducted in many studies, owing to bioethical concerns, despite it being the gold standard for diagnosis. Therefore, inconsistencies may occur regarding the definition of extra‐salivary gland complications.…”

Section: Discussionmentioning

confidence: 99%

The prevalence of extra‐salivary gland complications in immunoglobulin G4‐related sialadenitis

Zhou

Yan

et al. 2021

Oral Diseases

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Objectives:To estimate the prevalence of extra-glandular lesions in patients with immunoglobulin G4-related sialadenitis (IgG4-RS). Methods: Six electronic databases (PubMed, EMBASE, Science Direct, Scopus, Web of Science, and China National Knowledge Infrastructure) were systematically searched from the date of inception of each database to March 2021. The Strengthening the Reporting of Observational Studies in Epidemiology statement was used to conduct methodological quality assessment, and a random-effect meta-analysis model was applied to estimate the prevalence. Publication bias was visually assessed using a funnel plot and calculated via Begg's and Egger's tests. The Stata 15 software was used to perform data analysis.Results: A total of 43 articles comprising 1,864 patients with IgG4-RS were considered to be eligible for this study. The pooled prevalence of extra-salivary gland lesions in IgG4-RS was 76.53% with a confidence interval (CI) of (69.39%, 83.04%). A higher prevalence was associated with studies published before or during 2015 (84.38%, CI [74.23%, 92.58%]) than those published after 2015 (68.55%, CI [58.44%, 77.88%]). Lacrimal gland involvement (54.68%, CI [45.61%, 63.60%]) and lymph node swelling (56.96%, CI [48.16%, 65.56%]) were the most frequent lesions. Conclusions: Extra-glandular lesions were common in patients with IgG4-RS. More high-quality prospective studies with less heterogeneity are required to determine the accurate prevalence.

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Section: Discussionmentioning

confidence: 99%

The prevalence of extra‐salivary gland complications in immunoglobulin G4‐related sialadenitis

Zhou

Yan

et al. 2021

Oral Diseases

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“…Indeed, in the 2011 pathology consensus, biopsy samples in organs like lung, pancreas, bile duct, liver and kidney have lower IgG4/HPF threshold than that of surgical ones. While the cutoff of IgG4/HPF count for other organs including salivary gland, lacrimal gland, lymph node, pleura, retroperitoneum, aorta and skin suggested by the consensus, as we have observed, are more suitable to surgical samples but may be less applicable to the small biopsy [3,12] . It is noteworthy that setting the cutoff to IgG4/HPF > 10, surgical cases that meet this threshold are still signi cantly more than needle biopsies.…”

Section: Discussionmentioning

confidence: 99%

Needle Biopsy Compared With Surgical Biopsy: Pitfalls of Small Biopsy in Histologial Diagnosis of IgG4-related Disease

Yang

Chi

et al. 2020

Preprint

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ObjectiveThe growing utilization of needle biopsy has challenged the current pathology consensus of IgG4-related disease (IgG4-RD). The aims of this study were to identify the histological characteristics of needle biopsy and surgical specimens, and evaluate the ability of needle biopsy in histological diagnosis of IgG4-RD.MethodsBiopsies from patients who were referred to as IgG4-RD by the 2019 ACR/EULAR IgG4-RD classification criteria in Peking University People’s Hospital from 2012 to 2019 were re-evaluated. Typical histological features and diagnostic categories were compared between needle biopsy and surgical biopsy.ResultsIn total, 69 patients met the 2019 ACR/EULAR classification criteria and 72 biopsies of them were re-evaluated. All cases showed lymphoplasmacytic infiltrate, while storiform fibrosis and obliterative phlebitis were only present in 35 (48.6%) and 23 (31.9%) specimens, respectively. Storiform fibrosis was more likely to be seen in retroperitoneum lesion (P=0.033). Surgical biopsy showed significantly higher IgG4+ plasma cells/high power field (IgG4/HPF) count (P<0.01) and higher proportion of IgG4/HPF>10 (P<0.01). No significant difference was observed with regard to the ratio of IgG4+/IgG+ cells (IgG4/IgG) (P=0.399), storiform fibrosis (P=0.739), and obliterative phletibis (P=0.153). According to the 2011 comprehensive diagnostic criteria, patients who performed a needle biopsy were less likely to be probable IgG4-RD (P=0.045). Based on the 2011 pathology consensus, needle biopsy was tougher to be diagnosed as IgG4-RD (P<0.01), especially to be highly suggestive IgG4-RD (P<0.01). Only 1/18 (5.6%) needle salivary specimens fulfilled the cutoff of IgG4/HPF>100, which was significantly less than 15/23 (65.2%) of surgical ones (P<0.01).ConclusionsNeedle biopsy shows an inferiority in detecting IgG4/HPF count but not in IgG4/IgG ratio, storiform fibrosis and obliterative phlebitis. Compared with surgical samples, it is tougher for needle biopsy to obtain a histological diagnosis of IgG4-RD. A different IgG4/HPF threshold for needle biopsy of salivary glands may be considered.

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“…Based on the IgG4 staining, samples were classified as either IgG4-positive or IgG4-negative by counting the plasma cells immunopositive for IgG4 at three different HPFs (x40) in each specimen. We used the same cut-off point for IgG4-positivity as in our previous study: specimens with ≥70 IgG4positive plasma cells/HPF were categorized as IgG4-positive 5 and were included for further study and assessment of genuine IgG4-related disease. To do so, all IgG4-positive specimens were additionally immunostained for IgG, whereupon the IgG4/IgG ratio was calculated after determining the number of IgG-positive plasma cells at three HPFs.…”

Section: Evaluation Of Immunohistochemical Stainingmentioning

confidence: 99%

“…Among 51 patients diagnosed with chronic sclerosing sialadenitis, only two presented with genuine IgG4-related disease. Furthermore, these two specimens did not entirely fulfill the histopathological criteria for chronic sclerosing sialadenitis, but were instead classified as nonspecific sialadenitis 5 . Another significant finding was the presence of elevated numbers of IgG4-positive plasma cells in specimens associated with sialolithiasis and nonspecific forms of sialadenitis.…”

Section: Introductionmentioning

confidence: 99%

IgG4-positive plasma cells in nonspecific sialadenitis and sialolithiasis

et al. 2022

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Chronic sclerosing sialadenitis is commonly regarded as a manifestation of IgG4-related disease. We previously found that a high IgG4 expression or IgG4-related disease could accompany nonspecific sialadenitis, whereas chronic sclerosing sialadenitis was not directly associated with IgG4-related disease. Our previous findings lead us to hypothesize that these inflammatory conditions of the submandibular gland signify a continuous progression of disease rather than different disease entities. We, therefore, aimed to determine the presence of IgG4-positivity and genuine IgG4-related disease in a cohort of 165 submandibular gland specimens from patients who underwent surgery due to chronic nonspecific sialadenitis or sialolithiasis. To do so, we re-evaluated histopathological features and divided samples into three groups: (A) nonspecific sialadenitis without known sialolithiasis, (B) sialadenitis with sialolithiasis, and (C) sialolithiasis without sialadenitis. We performed immunohistochemical staining for IgG4, IgG, and CD31, and assessed the Boston consensus statement criteria for IgG4-related disease in IgG4-positive samples. We also reviewed patient records and supplemented follow-up data with a questionnaire among patients with IgG4-positive samples. IgG4-positive plasma cells (range 1–344) were found in 131 samples. Among these, 19 samples were classified as IgG4-positive (≥70 IgG4-positive plasma cells/high-power field). Two IgG4-positive samples were histologically highly suggestive of IgG4-related disease, but only one had a clinically confirmed diagnosis of IgG4-related disease. Our results indicate that patients with sialadenitis and sialolithiasis often present with IgG4-positive lymphoplasmacytic infiltrates, but exceedingly rarely present with genuine IgG4-related disease. In sialolithiasis without sialadenitis, IgG4-positive plasma cells are often absent or appear in small numbers. These results support our hypothesis of a continuum of disease, and indicate that progressive inflammation of the submandibular gland leads to the development of more specific pathological features over time.

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Sclerosing sialadenitis of the submandibular gland is rarely an immunoglobulin G4-related disease in the Finnish population

Cited by 14 publications

References 25 publications

The prevalence of extra‐salivary gland complications in immunoglobulin G4‐related sialadenitis

The prevalence of extra‐salivary gland complications in immunoglobulin G4‐related sialadenitis

Needle Biopsy Compared With Surgical Biopsy: Pitfalls of Small Biopsy in Histologial Diagnosis of IgG4-related Disease

IgG4-positive plasma cells in nonspecific sialadenitis and sialolithiasis

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