Cardiac arrhythmias as the initial manifestation of adult primary Sjögren's syndrome: a case report and literature review

Liang, Minrui; Bao, Liwen; Xiong, Nanqing; Jin, Bo; Ni, Hongbin; Zhang, Jinjin; Zou, Hejian; Luo, Xinping; Li, Jian

doi:10.1111/1756-185x.12616

Cited by 12 publications

(3 citation statements)

References 15 publications

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“…Fortunately, cardiac manifestations such as pericarditis, pulmonary hypertension and cardiomyopathy are very rare, affecting <1% of pSS patients, respectively [ 207 ]. Cardiac rhythm disturbances have been described, secondary to ionic disorders, dysautonomia or direct impairment of the electrical conduction system of the heart [ 224 , 225 ].…”

Section: Clinical Manifestationsmentioning

confidence: 99%

Current State of Knowledge on Primary Sjögren’s Syndrome, an Autoimmune Exocrinopathy

Parisis

Chivasso

Perret

et al. 2020

JCM

112

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Primary Sjögren’s syndrome (pSS) is a chronic systemic autoimmune rheumatic disease characterized by lymphoplasmacytic infiltration of the salivary and lacrimal glands, whereby sicca syndrome and/or systemic manifestations are the clinical hallmarks, associated with a particular autoantibody profile. pSS is the most frequent connective tissue disease after rheumatoid arthritis, affecting 0.3–3% of the population. Women are more prone to develop pSS than men, with a sex ratio of 9:1. Considered in the past as innocent collateral passive victims of autoimmunity, the epithelial cells of the salivary glands are now known to play an active role in the pathogenesis of the disease. The aetiology of the “autoimmune epithelitis” still remains unknown, but certainly involves genetic, environmental and hormonal factors. Later during the disease evolution, the subsequent chronic activation of B cells can lead to the development of systemic manifestations or non-Hodgkin’s lymphoma. The aim of the present comprehensive review is to provide the current state of knowledge on pSS. The review addresses the clinical manifestations and complications of the disease, the diagnostic workup, the pathogenic mechanisms and the therapeutic approaches.

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Section: Clinical Manifestationsmentioning

confidence: 99%

Current State of Knowledge on Primary Sjögren’s Syndrome, an Autoimmune Exocrinopathy

Parisis

Chivasso

Perret

et al. 2020

JCM

112

View full text Add to dashboard Cite

show abstract

“…We found that compared with the ICR mice, the submandibular gland tissue of NOD mice model had histology of acinar destruction, basement membrane changes, and lymphocyte infiltration. Studies have shown that SS, in addition to epithelial dysfunction, has several extraglandular manifestations associated with lymphocyte infiltration and B cell overactivity in other organs [40][41][42]. Numerous studies have shown that aquaporin AQP5 is a crucial target for determining salivary gland secretion and tissue formation [43][44][45].…”

Section: Discussionmentioning

confidence: 99%

The Critical Biomarkers Identification of Insulin Signaling Involved in Initiating cAMP Signaling Mediated Salivary Secretion in Sjogren Syndrome: Transcriptome Sequencing in NOD Mice Model

et al. 2022

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Background Sjogren’s syndrome (SS) is an autoimmune disorder characterized by the destruction of exocrine glands, resulting in dry mouth and eyes. Currently, there is no effective treatment for SS, and the mechanisms associated with inadequate salivary secretion are poorly understood. Methods In this study, we used NOD mice model to monitor changes in mice’s salivary secretion and water consumption. Tissue morphology of the submandibular glands was examined by H&E staining, and Immunohistochemical detected the expression of AQP5 (an essential protein in salivary secretion). Global gene expression profiling was performed on submandibular gland tissue of extracted NOD mice model using RNA-seq. Subsequently, a series of bioinformatics analyses of transcriptome sequencing was performed, including differentially expressed genes (DEGs) identification, Gene Ontology (GO) and Kyoto Encyclopedia of Genes and Genomes (KEGG) enrichment analysis, PPI network construction, hub gene identification, and the validity of diagnostic indicators using the dataset GSE40611. Finally, IFN-γ was used to treat the cells, the submandibular gland tissue of NOD mice model was extracted, and RT-qPCR was applied to verify the expression of hub genes. Results We found that NOD mice model had reduced salivary secretion and increased water consumption. H&E staining suggests acinar destruction and basement membrane changes in glandular tissue. Immunohistochemistry detects a decrease in AQP5 immunostaining within acinar. In transcriptome sequencing, 42 overlapping DEGs were identified, and hub genes (REN, A2M, SNCA, KLK3, TTR, and AZGP1) were identified as initiating targets for insulin signaling. In addition, insulin signaling and cAMP signaling are potential pathways for regulating salivary secretion and constructing a regulatory relationship between target-cAMP signaling-salivary secretion. Conclusion The new potential targets and signal axes for regulating salivary secretion provide a strategy for SS therapy in a clinical setting.

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“…160 Cardiac involvement is uncommon in pSS but atrioventricular block and complex cardiac arrhythmias have been described. 161,162 In one study, echocardiograms were performed in 107 consecutive subjects with SS and 112 matched healthy controls. 163 Among the SS cohort, the incidence of mitral, tricuspid, and aortic regurgitation was higher than that in controls (p < 0.001, p ¼ 0.02, and p < 0.007, respectively) and left ventricular mass index was increased in SS (p < 0.01).…”

Section: Cardiac and Cardiovascular Involvementmentioning

confidence: 99%

Pulmonary and Bronchiolar Involvement in Sjogren's Syndrome

Chung

Wilgus

Fishbein

et al. 2019

Semin Respir Crit Care Med

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Sjogren's syndrome (SS) is a chronic autoimmune disease characterized by mononuclear cells (principally lymphocytes) infiltrating exocrine glands (e.g., salivary and lacrimal glands), leading to destruction of exocrine epithelial cells and dryness of mucosal surfaces. Cardinal symptoms are dry eyes (xerophthalmia) and dry mouth (xerostomia). Extraglandular sites are affected in 30 to 40% of cases of SS (particularly neurological, kidneys, skin, and lungs). B cell hyperactivity, autoantibody production, and hypergammaglobulinemia are cardinal features of SS. Primary SS is not associated with other autoimmune diseases. However, SS can complicate diverse autoimmune disorders (particularly systemic lupus erythematosus, rheumatoid arthritis, and scleroderma); this form is termed “secondary SS.” Pulmonary involvement is usually not a dominant feature of SS, but may be severe in some cases. In this review, we discuss specific tracheal, bronchiolar, and pulmonary complications of SS including xerotrachea, bronchiolitis, bronchiectasis, interstitial lung disease, nonspecific interstitial pneumonia, usual interstitial pneumonia, lymphoid interstitial pneumonia, organizing pneumonia, acute fibrinous and organizing pneumonia, pulmonary cysts, pleural effusions, pulmonary amyloidosis, and bronchus- or lung-associated lymphomas.

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Cardiac arrhythmias as the initial manifestation of adult primary Sjögren's syndrome: a case report and literature review

Cited by 12 publications

References 15 publications

Current State of Knowledge on Primary Sjögren’s Syndrome, an Autoimmune Exocrinopathy

Current State of Knowledge on Primary Sjögren’s Syndrome, an Autoimmune Exocrinopathy

The Critical Biomarkers Identification of Insulin Signaling Involved in Initiating cAMP Signaling Mediated Salivary Secretion in Sjogren Syndrome: Transcriptome Sequencing in NOD Mice Model

Pulmonary and Bronchiolar Involvement in Sjogren's Syndrome

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