Abstract

To analyze the composition of the oral microbiome in children and adolescents with chronic nonbacterial osteomyelitis (CNO) with respect to age distribution, gender differences, effects of medication, disease activity and the influence of body site. The oral microbiome of 20patients (12male and 8female; median age 10.3years) and 36controls were examined. Two different sites of the oral cavity were swabbed at two time points. Current medication and disease activity were evaluated and registered at these time points. Samples were subjected to amplicon sequencing of the V4 region of the 16S rRNA gene and Qiime2 was used to calculate alpha and beta diversity for multiple alternative metrics. On the basis of relative abundances of 975 different suboperational taxonomic units in high throughput next generation sequencing, asignificant shift in the composition of the oral microbiome (p < 0.02) was observed among patients being treated with different medications. There was asignificant difference in bacterial communities between the group aged 3-8 years old and the group aged 9-14years old. Significant differences were also seen in bacterial colonization on different sites in the oral cavity, but not with respect to gender or disease activity. We present first data of apilot study of the oral microbiome in children and adolescents with CNO, arare autoinflammatory bone disease. Differences of the oral microbiome of diseased children to normal adult controls revealed apossible role of the oral microbiome as modulatory target or biomarker in CNO.

Highlights

  • Chronic nonbacterial osteomyelitis (CNO), known as chronic recurrent multifocal osteomyelitis (CRMO), is a disease spectrum encompassing a state of monofocal or multifocal autoinflammatory bone lesions, primarily affecting children and adolescents with an age peak between 7 and 12 years [1]

  • We present first data of a pilot study of the oral microbiome in children and adolescents with chronic nonbacterial osteomyelitis (CNO), a rare autoinflammatory bone disease

  • Differences of the oral microbiome of diseased children to normal adult controls revealed a possible role of the oral microbiome as modulatory target or biomarker in CNO

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Summary

Introduction

Chronic nonbacterial osteomyelitis (CNO), known as chronic recurrent multifocal osteomyelitis (CRMO), is a disease spectrum encompassing a state of monofocal or multifocal autoinflammatory bone lesions, primarily affecting children and adolescents with an age peak between 7 and 12 years [1]. In 2014, an interplay between microbial agents and bone erosions in an established CNO mouse model was discussed. In this mouse model, mice spontaneously develop osteomyelitis resembling CNO (Pstpip cmo mice). Diet-associated changes of the microbiome were found to be responsible for regulation of inflammation in an animal model of CNO [7]

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