Research Article: Dysbiotic salivary microbiota in dry mouth and primary Sjögren’s syndrome patients

Date Published: June 18, 2019

Publisher: Public Library of Science

Author(s): S. Rusthen, A. K. Kristoffersen, A. Young, H. K. Galtung, B. É. Petrovski, Ø. Palm, M. Enersen, J. L. Jensen, Silke Appel.


Primary Sjögren’s syndrome (pSS) is an autoimmune disease characterized by reduced lacrimal and salivary secretion. Sicca symptoms together with fatigue and musculoskeletal pain can significantly reduce the patients’ quality of life. Furthermore, low salivary secretion may disrupt the oral microbial homeostasis. The aim of this study was to compare the salivary microbiota from pSS patients with patients with sicca symptoms not fulfilling the classification criteria for pSS (non-SS), and with healthy controls without sicca complaints.

Pellets from centrifuged chewing-stimulated whole saliva from pSS patients (n = 15), non-SS sicca patients (n = 15) and healthy controls (n = 15) were prepared. DNA was extracted and analyzed by 16S rRNA gene sequencing. The acquired sequencing data were performed using the human oral microbiome database (HOMD).

We detected 42, 45, and 34 bacterial genera in saliva samples from pSS patients, non-SS sicca patients, and healthy controls, respectively. The most abundant genera in all samples were Prevotella, Veillonella, Streptococcus, and Haemophilus. At species level Streptococcus intermedius, Prevotella intermedia, Fusobacterium nucleatum subsp. vincentii, Porphyromonas endodontalis, Prevotella nancensis, Tannerella spp., and Treponema spp. were detected in the samples from pSS and non-SS only, while Porphyromonas pasteri was mostly found among the healthy controls.

Our study indicated dysbiosis in the salivary microbiota from pSS and non-SS patients compared to healthy controls. Additionally, the results showed that the salivary microbiome in the pSS group differed significantly from the non-SS group.

Partial Text

Sjögren’s syndrome (SS) is an autoimmune systemic inflammatory disease that affects exocrine glands, mainly the lacrimal and salivary glands. Lymphocytic infiltration of the gland results in destruction of the tissue, loss of function, and reduced secretion of tears and saliva. The etiology of SS remains to be elucidated, although both environmental and genetic factors are believed to be involved in the pathogenesis [1]. Clinical manifestations of SS include the classical sicca symptoms of dry eyes and dry mouth, together with fatigue and musculoskeletal pain [2]. Sjögren’s syndrome may present itself as primary SS (pSS) or secondary SS (sSS) when a connective tissue disease has been diagnosed prior to the development of sicca symptoms. In 2002, the American-European Consensus Group (AECG) proposed a set of classification criteria for pSS [3,4], that includes dry mouth, dry eyes, reduced salivary secretion, reduced lacrimal secretion, presence of Ro/SSA and/or La/SSB autoantibodies, and lymphocyte infiltration in minor salivary glands. In order to be classified as pSS, four of the six criteria must be met, including a positive minor salivary gland biopsy or positive serum antibodies. Alternatively, any three of the four objective criteria should be fulfilled. Interestingly, a serological profile characterized by anti-Ro/SSA and anti-La/SSB, antibodies against extractable nuclear antigens, has been reported in 50–70% and 25–40% of adults with pSS, respectively [5]. The prevalence of pSS is reported to vary from 0.05 to 1% in the European population, depending on which classification criteria have been used [6,7]. The criteria from the AECG are well accepted and are often used in research and clinical practice [8].

The study population consisted of 45 female participants aged 30 to 80 years that were divided into three groups of fifteen persons. The first group was composed of patients with pSS, who fulfilled the AECG classification criteria for pSS (pSS group). The second group consisted of subjects with sicca symptoms, but without anti-SSA/SSB autoantibodies and with a negative salivary gland biopsy, thus not fulfilling the AECG criteria for pSS (non-SS group). The third group was made up of healthy persons without complaints of dry mouth or dry eyes (control group).

In this study, the salivary bacterial profile of patients with pSS, dry mouth subjects (non-SS), and age-matched healthy controls was demonstrated using a 16S rRNA pyrosequencing approach. We found that the salivary bacterial profile of the pSS and non-SS groups differed from the controls. The analysis of the oral microflora at phylum level of the saliva samples showed the existence of nine bacterial phyla, including the same predominant phyla Firmicutes, Bacteroidetes, Actinobacteria, Proteobacteria, and Fusobacteria, that have been demonstrated in previous studies [9,14].




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