Evaluation of the Oral Bacterial Genome and Metabolites in Patients with Wolfram Syndrome

• Published in: International journal of molecular sciences Vol. 24; no. 6; p. 5596
• Main Authors: Zmysłowska-Polakowska, E, Płoszaj, T, Skoczylas, S, Mojsak, P, Ciborowski, M, Kretowski, A, Lukomska-Szymanska, M, Szadkowska, A, Mlynarski, W, Zmysłowska, A
• Format: Journal Article
• Language: English
• Published: Switzerland MDPI AG 15.03.2023; MDPI
• Subjects: Acetic acid; Alzheimer's disease; Amino acids; Analysis; Bacteria; Bacterial genetics; Benzoic acid; Campylobacter; Care and treatment; Diabetes; Diabetes mellitus; Diabetes mellitus (insulin dependent); Diabetes Mellitus, Type 1 - complications; Diabetes therapy; Evaluation; Gas chromatography; Genome, Bacterial; Genomes; Genomics; gingival samples; Humans; Instrument industry; Insulin; Lactic acid; Mass spectrometry; Mass spectroscopy; Metabolism; Metabolites; Metabolome; metabolomics; Microbiomes; Microbiota (Symbiotic organisms); Microorganisms; Nervous system diseases; Neurodegeneration; oral microbiome; Patients; RNA, Ribosomal, 16S - metabolism; rRNA 16S; Type 1 diabetes; Wolfram Syndrome; Poland; California; United States; Massachusetts; Taiwan; neurodegeneration; oral microbiome; Wolfram syndrome; gingival samples; metabolomics
• ISSN: 1422-0067; 1661-6596; 1422-0067
• DOI: 10.3390/ijms24065596

In Wolfram syndrome (WFS), due to the loss of wolframin function, there is increased ER stress and, as a result, progressive neurodegenerative disorders, accompanied by insulin-dependent diabetes. The aim of the study was to evaluate the oral microbiome and metabolome in WFS patients compared with patients with type 1 diabetes mellitus (T1DM) and controls. The buccal and gingival samples were collected from 12 WFS patients, 29 HbA1c-matched T1DM patients ( = 0.23), and 17 healthy individuals matched by age ( = 0.09) and gender ( = 0.91). The abundance of oral microbiota components was obtained by Illumina sequencing the 16S rRNA gene, and metabolite levels were measured by gas chromatography-mass spectrometry. (22.2%), (12.1%), and (10.8%) were the most common bacteria in the WFS patients, while comparisons between groups showed significantly higher abundance of , , , , and in the WFS group ( < 0.001). An ROC curve (AUC = 0.861) was constructed for the three metabolites that best discriminated WFS from T1DM and controls (acetic acid, benzoic acid, and lactic acid). Selected oral microorganisms and metabolites that distinguish WFS patients from T1DM patients and healthy individuals may suggest their possible role in modulating neurodegeneration and serve as potential biomarkers and indicators of future therapeutic strategies.