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European Journal of Pediatrics

, Volume 178, Issue 1, pp 89–95 | Cite as

The role of Cutibacterium acnes in auto-inflammatory bone disorders

  • Petra ZimmermannEmail author
  • Nigel CurtisEmail author
Original Article

Abstract

Chronic nonbacterial osteomyelitis (CNO) and SAPHO (synovitis, acne, pustulosis, hyperostosis and osteitis) syndrome are auto-inflammatory disorders manifesting as chronic inflammation of bones and joints, which in SAPHO is often accompanying by skin changes. The aetiology of these diseases is unknown, but includes genetic, infectious and immunological components. It has been proposed that Cutibacterium (formerly Propionibacterium) acnes plays a role in the pathogenesis. In this review, we summarise reported cases of CNO or SAPHO syndrome in which C. acnes has been isolated from bones. To identify cases, a search was done in May 2018 using the MEDLINE Ovid interface (1946 to present). We found 14 publications reporting 98 patients with auto-inflammatory bone disorders, of whom 48 (49%) had positive bone biopsies for C. acnes. This bacterium was more frequently isolated from open biopsies than percutaneous ones (43/69 (62%) vs 1/7 (14%); p = 0.04) and biopsies were more frequently positive in patients who presented with simultaneous skin manifestations (19/36 (53%) vs 4/12 (33%); p = 0.03).

Conclusion: In patients with CNO or SAPHO, C. acnes can be isolated from open biopsies suggesting that in these patients, C. acnes might be a pathogen rather than a contaminant. The fact that biopsies are more frequently positive in patients who present with simultaneous skin manifestations suggests that these individuals might have a genetic predisposition for impaired clearance of C. acnes.

What is known

Chronic nonbacterial osteomyelitis (CNO) and SAPHO (synovitis, acne, pustulosis, hyperostosis and osteitis) syndrome are auto-inflammatory disorders manifesting as inflammation of bones. Both diseases are an important differential diagnosis in children who present with symptoms of (multifocal) osteomyelitis.

The pathogenesis of CNO and SAPHO is multifactorial emcompassing genetic, infectious and immunological components, including interleukin (IL)-1 dysregulation. There is a controversy as to whether Cutibacterium (formerly Propionibacterium) acnes plays a role in the aetiology of CNO and SAPHO. It has been postulated that the presence of C. acnes might trigger auto-inflammatory chronic inflammation in genetically predisposed individuals.

What is new

In patients with CNO or SAPHO, C. acnes can be isolated more frequently from open biopsies, than from percutaneous ones, suggesting that C. acnes might be a pathogen rather than a contaminant.

Biopsies are more frequently positive in patients who present with simultaneous skin manifestations suggesting that these individuals might have a genetic predisposition for impaired clearance of C. acnes. Impaired C. acnes clearance likely leads to increased IL-1 beta (β) production by skin cells, bone cells and phagocytes, which is one of the main cytokines underlying chronic inflammatory bone disorders.

Keywords

SAPHO Spondyloarthopathies Chronic recurrent multifocal osteomyelitis CRMO DSOM Osteitis Hyperostosis Synovitis Pustulosis Osteomyelitis 

Abbreviations

C. acnes

Cutibacterium acnes

CNO

Chronic nonbacterial osteomyelitis

CRMO

Chronic recurrent multifocal osteomyelitis

DSOM

Diffuse sclerosing osteomyelitis of the mandible

IL

Interleukin

PPP

Palmoplantar pustulosis

SAPHO

Synovitis, acne, pustulosis, hyperostosis and osteitis

TNF

Tumour necrosis factor

Notes

Authors’ contributions

PZ drafted the initial manuscript and approved the final manuscript as submitted. NC critically reviewed and revised the manuscript and approved the final manuscript as submitted.

Compliance with ethical standards

Conflict of interest

The authors declare that there is no conflict of interest.

Research involving human participants and/or animals

N/A

Informed consent

N/A

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Copyright information

© Springer-Verlag GmbH Germany, part of Springer Nature 2018

Authors and Affiliations

  1. 1.Department of PaediatricsThe University of MelbourneParkvilleAustralia
  2. 2.Infectious Diseases UnitThe Royal Children’s Hospital MelbourneParkvilleAustralia
  3. 3.Infectious Diseases & Microbiology Research GroupMurdoch Children’s Research InstituteParkvilleAustralia
  4. 4.Infectious Diseases UnitUniversity of Basel Children’s HospitalBaselSwitzerland

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