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Published in: Pediatric Rheumatology 1/2018

Open Access 01-12-2018 | Research article

Chronic non bacterial osteitis- a multicentre study

Authors: Chandrika S. Bhat, Catriona Anderson, Aoibhinn Harbinson, Liza J. McCann, Marion Roderick, Adam Finn, Joyce E. Davidson, Athimalaipet V. Ramanan

Published in: Pediatric Rheumatology | Issue 1/2018

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Abstract

Objective

To understand the demographics, clinical features and treatment outcomes of Chronic Non-bacterial Osteitis (CNO) from three tertiary paediatric rheumatology services in the United Kingdom.

Methods

Children less than 18 years of age diagnosed with CNO between 2001 to 2016 from one tertiary service and between 2001 to 2017 from two tertiary services were included. Clinical notes were reviewed and all pertinent data were collected on a pre-defined proforma. One hundred and thirty one patients were included in the study. The Bristol diagnostic criteria were applied retrospectively.

Results

Retrospective analysis of the data showed that the disease was more common in girls than boys (2.5:1), median age at onset of symptoms was 9.5 years (IQR 8 to 11 years). Bone pain was the predominant symptom in 118/129 (91.4%) followed by swelling in 50/102 (49.01%). Raised inflammatory markers were present in 39.68% of the patients. Whole body Magnetic Resonance Imaging (MRI) was a useful diagnostic tool. Metaphyses of long bones were most often involved and the distal tibial metaphyses 65/131 (49.6%) was the most common site. Non-steroidal anti-inflammatory drugs were used as first line (81.67%) followed by bisphosphonates (61.79%). Treatment was escalated to a TNF blocker when response to bisphosphonates was suboptimal. The disease was in remission in 82.4% of the patients during the last follow up.

Conclusion

Our multicentre study describes features and outcomes of CNO in a large number of patients in the United Kingdom.

Significance and innovation

  • Raised inflammatory markers were present in 39.68% of our patients.
  • Whole body MRI is useful for diagnosis and also determining response to treatment.
  • A greater number of lesions were detected on radiological imaging compared to clinical assessment.
  • Metaphyses of long bones were most often involved and the distal tibial metaphyses (49.6%) were the most common site.
  • Non-steroidal anti-inflammatory drugs were used as first line (81.67%) followed by bisphosphonates (61.79%).
  • There was no difference in number of medications used for management in unifocal versus multifocal disease.
  • TNF blockers were used with good effect in our cohort.
Literature
1.
go back to reference Hedrich CM, Hofmann SR, Pablik J, Morbach H, Girschick HJ. Autoinflammatory bone disorders with special focus on chronic recurrent multifocal osteomyelitis (CRMO). Pediatr Rheumatol Online J. 2013;11(1):47.CrossRef Hedrich CM, Hofmann SR, Pablik J, Morbach H, Girschick HJ. Autoinflammatory bone disorders with special focus on chronic recurrent multifocal osteomyelitis (CRMO). Pediatr Rheumatol Online J. 2013;11(1):47.CrossRef
2.
go back to reference Ferguson PJ, Sandu M. Current understanding of the pathogenesis and management of chronic recurrent multifocal osteomyelitis. Curr Rheumatol Rep. 2012;14(2):130–41.CrossRef Ferguson PJ, Sandu M. Current understanding of the pathogenesis and management of chronic recurrent multifocal osteomyelitis. Curr Rheumatol Rep. 2012;14(2):130–41.CrossRef
3.
go back to reference Girschick HJ, Raab P, Surbaum S, Trusen A, Kirschner S, Schneider P, et al. Chronic non-bacterial osteomyelitis in children. Ann Rheum Dis. 2005;64(2):279–85.CrossRef Girschick HJ, Raab P, Surbaum S, Trusen A, Kirschner S, Schneider P, et al. Chronic non-bacterial osteomyelitis in children. Ann Rheum Dis. 2005;64(2):279–85.CrossRef
4.
go back to reference Beretta-Piccoli BC, Sauvain MJ, Gal I, Schibler A, Saurenmann T, Kressebuch H, et al. Synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome in childhood: a report of ten cases and review of the literature. Eur J Pediatr. 2000;159(8):594–601.CrossRef Beretta-Piccoli BC, Sauvain MJ, Gal I, Schibler A, Saurenmann T, Kressebuch H, et al. Synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome in childhood: a report of ten cases and review of the literature. Eur J Pediatr. 2000;159(8):594–601.CrossRef
5.
go back to reference Cox AJ, Darbro BW, Laxer RM, Velez G, Bing X, Finer AL, et al. Recessive coding and regulatory mutations in FBLIM1 underlie the pathogenesis of chronic recurrent multifocal osteomyelitis (CRMO). PLoS One. 2017;12(3):e0169687.CrossRef Cox AJ, Darbro BW, Laxer RM, Velez G, Bing X, Finer AL, et al. Recessive coding and regulatory mutations in FBLIM1 underlie the pathogenesis of chronic recurrent multifocal osteomyelitis (CRMO). PLoS One. 2017;12(3):e0169687.CrossRef
6.
go back to reference Girschick H, Finetti M, Orlando F, Schalm S, Insalaco A, Ganser G, et al. The multifaceted presentation of chronic recurrent multifocal osteomyelitis: a series of 486 cases from the Eurofever international registry. Rheumatology (Oxford). 2018;57(8):1504.CrossRef Girschick H, Finetti M, Orlando F, Schalm S, Insalaco A, Ganser G, et al. The multifaceted presentation of chronic recurrent multifocal osteomyelitis: a series of 486 cases from the Eurofever international registry. Rheumatology (Oxford). 2018;57(8):1504.CrossRef
7.
go back to reference Catalano-Pons C, Comte A, Wipff J, Quartier P, Faye A, Gendrel D, et al. Clinical outcome in children with chronic recurrent multifocal osteomyelitis. Rheumatology (Oxford). 2008;47(9):1397–9.CrossRef Catalano-Pons C, Comte A, Wipff J, Quartier P, Faye A, Gendrel D, et al. Clinical outcome in children with chronic recurrent multifocal osteomyelitis. Rheumatology (Oxford). 2008;47(9):1397–9.CrossRef
8.
go back to reference Jansson A, Renner ED, Ramser J, Mayer A, Haban M, Meindl A, et al. Classification of non-bacterial osteitis: retrospective study of clinical, immunological and genetic aspects in 89 patients. Rheumatology (Oxford). 2007;46(1):154–60.CrossRef Jansson A, Renner ED, Ramser J, Mayer A, Haban M, Meindl A, et al. Classification of non-bacterial osteitis: retrospective study of clinical, immunological and genetic aspects in 89 patients. Rheumatology (Oxford). 2007;46(1):154–60.CrossRef
9.
go back to reference Wipff J, Costantino F, Lemelle I, Pajot C, Duquesne A, Lorrot M, et al. A large national cohort of French patients with chronic recurrent multifocal osteitis. Arthritis Rheumatol. 2015;67(4):1128–37.CrossRef Wipff J, Costantino F, Lemelle I, Pajot C, Duquesne A, Lorrot M, et al. A large national cohort of French patients with chronic recurrent multifocal osteitis. Arthritis Rheumatol. 2015;67(4):1128–37.CrossRef
10.
go back to reference Roderick MR, Ramanan AV. Chronic recurrent multifocal osteomyelitis. Adv Exp Med Biol. 2013;764:99–107.CrossRef Roderick MR, Ramanan AV. Chronic recurrent multifocal osteomyelitis. Adv Exp Med Biol. 2013;764:99–107.CrossRef
11.
go back to reference Vittecoq O, Said LA, Michot C, Mejjad O, Thomine JM, Mitrofanoff P, et al. Evolution of chronic recurrent multifocal osteitis toward spondylarthropathy over the long term. Arthritis Rheum. 2000;43(1):109–19.CrossRef Vittecoq O, Said LA, Michot C, Mejjad O, Thomine JM, Mitrofanoff P, et al. Evolution of chronic recurrent multifocal osteitis toward spondylarthropathy over the long term. Arthritis Rheum. 2000;43(1):109–19.CrossRef
12.
go back to reference Jansson AF, Müller TH, Gliera L, Ankerst DP, Wintergerst U, Belohradsky BH, et al. Clinical score for nonbacterial osteitis in children and adults. Arthritis Rheum. 2009;60(4):1152–9.CrossRef Jansson AF, Müller TH, Gliera L, Ankerst DP, Wintergerst U, Belohradsky BH, et al. Clinical score for nonbacterial osteitis in children and adults. Arthritis Rheum. 2009;60(4):1152–9.CrossRef
13.
go back to reference Zhao Y, Dedeoglu F, Ferguson PJ, Lapidus SK, Laxer RM, Bradford MC, et al. Physicians' perspectives on the diagnosis and treatment of chronic nonbacterial osteomyelitis. Int J Rheumatol. 2017;2017:7694942.CrossRef Zhao Y, Dedeoglu F, Ferguson PJ, Lapidus SK, Laxer RM, Bradford MC, et al. Physicians' perspectives on the diagnosis and treatment of chronic nonbacterial osteomyelitis. Int J Rheumatol. 2017;2017:7694942.CrossRef
14.
go back to reference Zhao Y, Wu EY, Oliver MS, Cooper AM, Basiaga ML, Vora SS, et al. Consensus treatment plans for chronic nonbacterial osteomyelitis refractory to nonsteroidal anti-inflammatory drugs and/or with active spinal lesions. Arthritis Care Res (Hoboken). 2018;70(8):1228–1237.CrossRef Zhao Y, Wu EY, Oliver MS, Cooper AM, Basiaga ML, Vora SS, et al. Consensus treatment plans for chronic nonbacterial osteomyelitis refractory to nonsteroidal anti-inflammatory drugs and/or with active spinal lesions. Arthritis Care Res (Hoboken). 2018;70(8):1228–1237.CrossRef
15.
go back to reference Walsh P, Manners PJ, Vercoe J, Burgner D, Murray KJ. Chronic recurrent multifocal osteomyelitis in children: nine years' experience at a statewide tertiary paediatric rheumatology referral Centre. Rheumatology (Oxford). 2015;54(9):1688–91.CrossRef Walsh P, Manners PJ, Vercoe J, Burgner D, Murray KJ. Chronic recurrent multifocal osteomyelitis in children: nine years' experience at a statewide tertiary paediatric rheumatology referral Centre. Rheumatology (Oxford). 2015;54(9):1688–91.CrossRef
Metadata
Title
Chronic non bacterial osteitis- a multicentre study
Authors
Chandrika S. Bhat
Catriona Anderson
Aoibhinn Harbinson
Liza J. McCann
Marion Roderick
Adam Finn
Joyce E. Davidson
Athimalaipet V. Ramanan
Publication date
01-12-2018
Publisher
BioMed Central
Published in
Pediatric Rheumatology / Issue 1/2018
Electronic ISSN: 1546-0096
DOI
https://doi.org/10.1186/s12969-018-0290-5

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