A longitudinal cohort study of Finnish patients with primary Sjogren's syndrome: clinical, immunological, and epidemiological aspects

Pertovaara, Marja; Pukkala, Eero; Laippala, Pekka; Miettinen, A; Pasternack, Amos

doi:10.1136/ard.60.5.467

Cited by 154 publications

(89 citation statements)

References 35 publications

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“…The total observation time was 3,648 patient-years at risk. Our results confirm the findings of earlier smaller studies (8)(9)(10) and the recently published large retrospective study from 2 university hospital patient cohorts in Greece (11), i.e., a statistically significant increase in all-cause mortality among patients with primary SS could not be documented. We used the new AECC for assessing whether patients with autoimmune disease have worse long-term outcome than those with sicca symptoms without detectable autoimmune disturbances or more nonspecific immune activation.…”

Section: Discussionsupporting

confidence: 91%

“…Martens et al (8) studied 50 patients with primary SS at the Mayo Clinic in Rochester, Minnesota and reported survival of these patients to be comparable with that of the healthy population. In a recent study in Finland, the standardized mortality ratio (SMR) in 110 patients with primary SS was 1.2; 70% of these patients had classic focal sialadenitis (focus score Ն1) seen on salivary gland biopsy (9). In a study of 261 Greek patients, Skopouli et al found an SMR of 2.07 (95% confidence interval [95% CI] 1.03-3.71), with a low C4 level at diagnosis as a predictor variable for death (10).…”

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confidence: 99%

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Mortality and causes of death in primary Sjögren's syndrome: A prospective cohort study

Theander

Manthorpe

Jacobsson

2004

Arthritis & Rheumatism

235

125

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Objective.This study was undertaken to analyze standardized mortality ratios (SMRs) and causes and predictors of death in primary Sjögren's syndrome (SS) diagnosed according to 3 different classification criteria sets (the Copenhagen criteria, the European criteria, and the American-European consensus criteria (AECC).Methods. A linked registry study using information from the Malmö Primary SS Registry combined with the Swedish Cause-of-Death Registry was performed, and SMRs were calculated. Kaplan-Meier survival curves and log rank tests were used to compare survival probability between subgroups of patients with primary SS. Cox regression analysis was used to study the predictive value of various laboratory findings at the time of diagnosis.Results. Four hundred eighty-four patients with a median followup of 7 years (range 1 month to 17 years 11 months) were included. The SMR for those fulfilling the AECC (n ‫؍‬ 265) was 1.17 (95% confidence interval [95% CI] 0.81-1.63). Thirty-four deaths occurred in this group of patients. Excess mortality was found only for lymphoproliferative malignancy (cause-specific SMR 7.89 [95% CI 2.89-17.18]), corresponding to 2.53 excess deaths per 1,000 person-years at risk. In those not fulfilling the AECC (n ‫؍‬ 219), 14 deaths occurred, the SMR was 0.71 (95% CI 0.39-1.20), and no excess mortality due to any specific cause was found. Hypocomplementemia, defined as C3 and/or C4 values in the lowest quartile of the SS patients' values at the time of diagnosis, was a significant predictor of death, mainly due to lymphoproliferative malignancy.Conclusion. No increased all-cause mortality could be detected for patients with primary SS compared with the general population. When subgroups of primary SS were compared, excess mortality due to lymphoproliferative malignancy was found in patients fulfilling the AECC, the strongest predictor for unfavorable outcome being low C3 and/or C4 levels at the time of diagnosis.

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Section: Discussionsupporting

confidence: 91%

mentioning

confidence: 99%

Mortality and causes of death in primary Sjögren's syndrome: A prospective cohort study

Theander

Manthorpe

Jacobsson

2004

Arthritis & Rheumatism

235

125

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“…Only consecutive patients with primary SS who fulfilled the American-European Consensus Group criteria were recruited, and the study population was representative of the disease with regard to clinical, biologic, and immunologic features (26).…”

Section: Discussionmentioning

confidence: 99%

Association of transforming growth factor β1 and tumor necrosis factor α polymorphisms with anti‐SSB/La antibody secretion in patients with primary Sjögren's syndrome

Gottenberg¹,

Busson²,

Loiseau³

et al. 2004

Arthritis & Rheumatism

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Objective. To determine whether cytokine gene polymorphisms of interferon-␥ (IFN␥), interleukin-6 (IL-6), IL-10, tumor necrosis factor ␣ (TNF␣), and transforming growth factor ␤1 (TGF␤1) predispose subjects to the development of primary Sjögren's syndrome (SS).Methods. Single-base-exchange cytokine gene polymorphisms were analyzed in 129 French patients with primary SS who fulfilled the American-European Consensus Group criteria, as well as in 96 unrelated healthy subjects.Results. The frequency of the TNF-308A (TNF2) allele was significantly higher in the SS patients (26% versus 11%). This TNF2 association was restricted to patients with anti-SSB (37% versus 11% in controls). Stratification did not reveal an independent effect of TNF2 and HLA-DRB1*03 on disease or on anti-SSB antibody secretion. The frequency of allele C at codon 10 of TGF␤1 was strongly increased in the subgroup of patients with anti-SSB; this allele acted synergistically with DRB1*03 to predispose patients to the secretion of anti-SSB. The IL-10 GCC haplotype carrier rate was significantly higher in SS patients than in controls (67% versus 48%), but the IL-10 allele and genotype frequencies were not significantly different. No association was found between IL-6 or IFN␥ polymorphisms and primary SS.Conclusion. TNF2 was associated with anti-SSB antibody secretion, although this association was not independent of the association with DRB1*03. Allele C at codon 10 of TGF␤1 was found to act synergistically with DRB1*03 in predisposing patients to the secretion of anti-SSB. These results therefore suggest that most of the known genetic predisposition to primary SS might concern the pattern of autoantibody diversification.

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“…Among these, Hashimoto's thyroiditis (HT) and Sjögren's syndrome (SS) are the best examples with increased relative risks of 3-67 43,44 and 9-44, 43,[45][46][47][48] respectively to develop extranodal marginal zone B-cell lymphomas (MZBCLs). Moreover, for SS patients a 9-fold increased risk of obtaining a diffuse large B-cell lymphoma (DLBCL) has been reported.…”

Section: Auto-immune Inflammatory Conditions Associated With B-cell Lmentioning

confidence: 99%

Chronic inflammatory disease, lymphoid tissue neogenesis and extranodal marginal zone B-cell lymphomas

Bende¹,

Maldegem²,

Noesel³

2009

Haematologica

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Chronic autoimmune or pathogen-induced immune reactions resulting in lymphoid neogenesis are associated with development of malignant lymphomas, mostly extranodal marginal zone B-cell lymphomas (MZBCLs). In this review we address (i) chemokines and adhesion molecules involved in lymphoid neogenesis; (ii) the autoimmune diseases and pathogens which are associated with development of B-cell lymphomas; (iii) the molecular mechanisms involved in the initiation and progression of MZBCL; and (iv) 'potential' mouse models for MZBCL.Key words: B-cell non-Hodgkin's lymphoma, extranodal marginal zone B-cell lymphoma, immunoglobulin, B-cell antigen receptor, inflammation, lymphoid tissue neogenesis. Haematologica 2009;94:1109-1123. doi:10.3324/haematol.2009 This is an open-access paper. Citation: Bende RJ, van Maldegem F, and van Noesel CJM. Chronic inflammatory disease, lymphoid tissue neogenesis and extranodal marginal zone B cell lymphomas. ABSTRACT Chronic inflammatory disease, lymphoid tissue neogenesis and extranodal marginal zone B-cell lymphomas

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A longitudinal cohort study of Finnish patients with primary Sjogren's syndrome: clinical, immunological, and epidemiological aspects

Cited by 154 publications

References 35 publications

Mortality and causes of death in primary Sjögren's syndrome: A prospective cohort study

Mortality and causes of death in primary Sjögren's syndrome: A prospective cohort study

Association of transforming growth factor β1 and tumor necrosis factor α polymorphisms with anti‐SSB/La antibody secretion in patients with primary Sjögren's syndrome

Chronic inflammatory disease, lymphoid tissue neogenesis and extranodal marginal zone B-cell lymphomas

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