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The role of Cutibacterium acnes in auto-inflammatory bone disorders.
Eur J Pediatr 2019; 178(1):89-95EJ

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.

Authors+Show Affiliations

Department of Paediatrics, The University of Melbourne, Parkville, Australia. petra.zimmermann@mcri.edu.au. Infectious Diseases Unit, The Royal Children's Hospital Melbourne, Parkville, 3052, Australia. petra.zimmermann@mcri.edu.au. Infectious Diseases & Microbiology Research Group, Murdoch Children's Research Institute, Parkville, Australia. petra.zimmermann@mcri.edu.au. Infectious Diseases Unit, University of Basel Children's Hospital, Basel, Switzerland. petra.zimmermann@mcri.edu.au.Department of Paediatrics, The University of Melbourne, Parkville, Australia. nigel.curtis@rch.org.au. Infectious Diseases Unit, The Royal Children's Hospital Melbourne, Parkville, 3052, Australia. nigel.curtis@rch.org.au. Infectious Diseases & Microbiology Research Group, Murdoch Children's Research Institute, Parkville, Australia. nigel.curtis@rch.org.au.

Pub Type(s)

Journal Article
Review

Language

eng

PubMed ID

30324232

Citation

Zimmermann, Petra, and Nigel Curtis. "The Role of Cutibacterium Acnes in Auto-inflammatory Bone Disorders." European Journal of Pediatrics, vol. 178, no. 1, 2019, pp. 89-95.
Zimmermann P, Curtis N. The role of Cutibacterium acnes in auto-inflammatory bone disorders. Eur J Pediatr. 2019;178(1):89-95.
Zimmermann, P., & Curtis, N. (2019). The role of Cutibacterium acnes in auto-inflammatory bone disorders. European Journal of Pediatrics, 178(1), pp. 89-95. doi:10.1007/s00431-018-3263-2.
Zimmermann P, Curtis N. The Role of Cutibacterium Acnes in Auto-inflammatory Bone Disorders. Eur J Pediatr. 2019;178(1):89-95. PubMed PMID: 30324232.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - The role of Cutibacterium acnes in auto-inflammatory bone disorders. AU - Zimmermann,Petra, AU - Curtis,Nigel, Y1 - 2018/10/15/ PY - 2018/07/06/received PY - 2018/09/24/accepted PY - 2018/09/10/revised PY - 2018/10/17/pubmed PY - 2019/1/29/medline PY - 2018/10/17/entrez KW - CRMO KW - Chronic recurrent multifocal osteomyelitis KW - DSOM KW - Hyperostosis KW - Osteitis KW - Osteomyelitis KW - Pustulosis KW - SAPHO KW - Spondyloarthopathies KW - Synovitis SP - 89 EP - 95 JF - European journal of pediatrics JO - Eur. J. Pediatr. VL - 178 IS - 1 N2 - 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. SN - 1432-1076 UR - https://www.unboundmedicine.com/medline/citation/30324232/The_role_of_Cutibacterium_acnes_in_auto-inflammatory_bone_disorders L2 - https://dx.doi.org/10.1007/s00431-018-3263-2 DB - PRIME DP - Unbound Medicine ER -