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Published in: Pediatric Rheumatology 1/2019

Open Access 01-12-2019 | Bisphosphonate | Research article

Chronic non-bacterial osteomyelitis: a comparative study between children and adults

Authors: Andrea Skrabl-Baumgartner, Peter Singer, Theresa Greimel, Gregor Gorkiewicz, Josef Hermann

Published in: Pediatric Rheumatology | Issue 1/2019

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Abstract

Background

To compare clinical presentation, diagnostic and treatment strategies, and outcome between pediatric and adult patients with chronic non-bacterial osteomyelitis (CNO).

Methods

Retrospective single-centre comparative study of pediatric and adult patients diagnosed with chronic recurrent multifocal osteomyelitis (CRMO)/CNO or synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome treated at the Medical University of Graz.

Results

24 pediatric patients diagnosed with CRMO/CNO and 10 adult patients diagnosed with SAPHO syndrome were compared. Median age at diagnosis was 12.3 years (range 7.9–18.9) in the pediatric group and 32.5 years (range 22–56) in the adult group. Median time to diagnosis was shorter in children than in adults (0.3 vs. 1.0 years). Initial clinical presentation, laboratory and histopathological findings were similar in children and adults. Mean numbers of bone lesions were comparable between pediatric and adult patients (3.1 vs. 3.0), as were rates of skin involvement (33% vs. 30%). Sternal involvement was more frequent in adults whereas involvement of clavicle and long bones was more frequent in children (41.7% vs.10, 33% vs. 10%). Computerized tomography (CT) was used more often in adults, whereas whole-body magnetic resonance imaging (MRI) was used only in children. Bisphosphonates were applied more often in children and outcome was better in children than in adults (62.5% vs.30%).

Conclusion

Results of our study suggest that CNO/CRMO and SAPHO syndrome in children and adults might represent a single clinical syndrome that needs a similar diagnostic and therapeutic approach.
Literature
1.
go back to reference Giedion A, Holthusen W, Masel LF, Vischer D. Subacute and chronic "symmetrical" osteomyelitis. Ann Radiol (Paris). 1972;15:329–42. Giedion A, Holthusen W, Masel LF, Vischer D. Subacute and chronic "symmetrical" osteomyelitis. Ann Radiol (Paris). 1972;15:329–42.
2.
go back to reference Bjorksten B, Gustavson KH, Eriksson B, Lindholm A, Nordstrom S. Chronic recurrent multifocal osteomyelitis and pustulosis palmoplantaris. J Pediatr. 1978;93:227–31.CrossRef Bjorksten B, Gustavson KH, Eriksson B, Lindholm A, Nordstrom S. Chronic recurrent multifocal osteomyelitis and pustulosis palmoplantaris. J Pediatr. 1978;93:227–31.CrossRef
3.
go back to reference Bousvaros A, Marcon M, Treem W, Waters P, Issenman R, Couper R, et al. Chronic recurrent multifocal osteomyelitis associated with chronic inflammatory bowel disease in children. Dig Dis Sci. 1999;44:2500–7.CrossRef Bousvaros A, Marcon M, Treem W, Waters P, Issenman R, Couper R, et al. Chronic recurrent multifocal osteomyelitis associated with chronic inflammatory bowel disease in children. Dig Dis Sci. 1999;44:2500–7.CrossRef
4.
go back to reference Morbach H, Dick A, Beck C, Stenzel M, Muller-Hermelink HK, Raab P, et al. Association of chronic non-bacterial osteomyelitis with crohn's disease but not with card15 gene variants. Rheumatol Int. 2010;30:617–21.CrossRef Morbach H, Dick A, Beck C, Stenzel M, Muller-Hermelink HK, Raab P, et al. Association of chronic non-bacterial osteomyelitis with crohn's disease but not with card15 gene variants. Rheumatol Int. 2010;30:617–21.CrossRef
5.
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. 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.
6.
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;64:279–85.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;64:279–85.CrossRef
7.
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;46:154–60.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;46:154–60.CrossRef
8.
go back to reference Chamot AM, Benhamou CL, Kahn MF, Beraneck L, Kaplan G, Prost A. Acne-pustulosis-hyperostosis-osteitis syndrome. Results of a national survey. 85 cases. Rev Rhum Mal Osteoartic. 1987;54:187–96.PubMed Chamot AM, Benhamou CL, Kahn MF, Beraneck L, Kaplan G, Prost A. Acne-pustulosis-hyperostosis-osteitis syndrome. Results of a national survey. 85 cases. Rev Rhum Mal Osteoartic. 1987;54:187–96.PubMed
9.
go back to reference Greenwood S, Leone A, Cassar-Pullicino VN. Sapho and recurrent multifocal osteomyelitis. Radiol Clin N Am. 2017;55:1035–53.CrossRef Greenwood S, Leone A, Cassar-Pullicino VN. Sapho and recurrent multifocal osteomyelitis. Radiol Clin N Am. 2017;55:1035–53.CrossRef
10.
go back to reference Braun J, Bollow M, Remlinger G, Eggens U, Rudwaleit M, Distler A, et al. Prevalence of spondylarthropathies in hla-b27 positive and negative blood donors. Arthritis Rheum. 1998;41:58–67.CrossRef Braun J, Bollow M, Remlinger G, Eggens U, Rudwaleit M, Distler A, et al. Prevalence of spondylarthropathies in hla-b27 positive and negative blood donors. Arthritis Rheum. 1998;41:58–67.CrossRef
11.
go back to reference Schnabel A, Range U, Hahn G, Berner R, Hedrich CM. Treatment response and longterm outcomes in children with chronic nonbacterial osteomyelitis. J Rheumatol. 2017;44:1058–65.CrossRef Schnabel A, Range U, Hahn G, Berner R, Hedrich CM. Treatment response and longterm outcomes in children with chronic nonbacterial osteomyelitis. J Rheumatol. 2017;44:1058–65.CrossRef
12.
go back to reference Rudwaleit M, Landewe R, van der Heijde D, Listing J, Brandt J, Braun J, et al. The development of assessment of spondyloarthritis international society classification criteria for axial spondyloarthritis (part i): classification of paper patients by expert opinion including uncertainty appraisal. Ann Rheum Dis. 2009;68:770–6.CrossRef Rudwaleit M, Landewe R, van der Heijde D, Listing J, Brandt J, Braun J, et al. The development of assessment of spondyloarthritis international society classification criteria for axial spondyloarthritis (part i): classification of paper patients by expert opinion including uncertainty appraisal. Ann Rheum Dis. 2009;68:770–6.CrossRef
13.
go back to reference Kaiser D, Bolt I, Hofer M, Relly C, Berthet G, Bolz D, et al. Chronic nonbacterial osteomyelitis in children: a retrospective multicenter study. Pediatr Rheumatol Online J. 2015;13:25.CrossRef Kaiser D, Bolt I, Hofer M, Relly C, Berthet G, Bolz D, et al. Chronic nonbacterial osteomyelitis in children: a retrospective multicenter study. Pediatr Rheumatol Online J. 2015;13:25.CrossRef
14.
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;43:109–19.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;43:109–19.CrossRef
15.
go back to reference Hayem G, Bouchaud-Chabot A, Benali K, Roux S, Palazzo E, Silbermann-Hoffman O, et al. Sapho syndrome: a long-term follow-up study of 120 cases. Semin Arthritis Rheum. 1999;29:159–71.CrossRef Hayem G, Bouchaud-Chabot A, Benali K, Roux S, Palazzo E, Silbermann-Hoffman O, et al. Sapho syndrome: a long-term follow-up study of 120 cases. Semin Arthritis Rheum. 1999;29:159–71.CrossRef
16.
go back to reference Depasquale R, Kumar N, Lalam RK, Tins BJ, Tyrrell PN, Singh J, et al. Sapho: what radiologists should know. Clin Radiol. 2012;67:195–206.CrossRef Depasquale R, Kumar N, Lalam RK, Tins BJ, Tyrrell PN, Singh J, et al. Sapho: what radiologists should know. Clin Radiol. 2012;67:195–206.CrossRef
17.
go back to reference Morbach H, Schneider P, Schwarz T, Hofmann C, Raab P, Neubauer H, et al. Comparison of magnetic resonance imaging and 99mtechnetium-labelled methylene diphosphonate bone scintigraphy in the initial assessment of chronic non-bacterial osteomyelitis of childhood and adolescents. Clin Exp Rheumatol. 2012;30:578–82.PubMed Morbach H, Schneider P, Schwarz T, Hofmann C, Raab P, Neubauer H, et al. Comparison of magnetic resonance imaging and 99mtechnetium-labelled methylene diphosphonate bone scintigraphy in the initial assessment of chronic non-bacterial osteomyelitis of childhood and adolescents. Clin Exp Rheumatol. 2012;30:578–82.PubMed
18.
go back to reference Fritz J, Tzaribatchev N, Claussen CD, Carrino JA, Horger MS. Chronic recurrent multifocal osteomyelitis: comparison of whole-body mr imaging with radiography and correlation with clinical and laboratory data. Radiology. 2009;252:842–51.CrossRef Fritz J, Tzaribatchev N, Claussen CD, Carrino JA, Horger MS. Chronic recurrent multifocal osteomyelitis: comparison of whole-body mr imaging with radiography and correlation with clinical and laboratory data. Radiology. 2009;252:842–51.CrossRef
19.
go back to reference Salles M, Olive A, Perez-Andres R, Holgado S, Mateo L, Riera E, et al. The sapho syndrome: a clinical and imaging study. Clin Rheumatol. 2011;30:245–9.CrossRef Salles M, Olive A, Perez-Andres R, Holgado S, Mateo L, Riera E, et al. The sapho syndrome: a clinical and imaging study. Clin Rheumatol. 2011;30:245–9.CrossRef
20.
go back to reference Hospach T, Langendoerfer M, von Kalle T, Maier J, Dannecker GE. Spinal involvement in chronic recurrent multifocal osteomyelitis (crmo) in childhood and effect of pamidronate. Eur J Pediatr. 2010;169:1105–11.CrossRef Hospach T, Langendoerfer M, von Kalle T, Maier J, Dannecker GE. Spinal involvement in chronic recurrent multifocal osteomyelitis (crmo) in childhood and effect of pamidronate. Eur J Pediatr. 2010;169:1105–11.CrossRef
21.
go back to reference Gleeson H, Wiltshire E, Briody J, Hall J, Chaitow J, Sillence D, et al. Childhood chronic recurrent multifocal osteomyelitis: Pamidronate therapy decreases pain and improves vertebral shape. J Rheumatol. 2008;35:707–12.PubMed Gleeson H, Wiltshire E, Briody J, Hall J, Chaitow J, Sillence D, et al. Childhood chronic recurrent multifocal osteomyelitis: Pamidronate therapy decreases pain and improves vertebral shape. J Rheumatol. 2008;35:707–12.PubMed
22.
go back to reference Hofmann SR, Schnabel A, Rosen-Wolff A, Morbach H, Girschick HJ, Hedrich CM. Chronic nonbacterial osteomyelitis: pathophysiological concepts and current treatment strategies. J Rheumatol. 2016;43:1956–64.CrossRef Hofmann SR, Schnabel A, Rosen-Wolff A, Morbach H, Girschick HJ, Hedrich CM. Chronic nonbacterial osteomyelitis: pathophysiological concepts and current treatment strategies. J Rheumatol. 2016;43:1956–64.CrossRef
23.
go back to reference Pearce MS, Salotti JA, Little MP, McHugh K. L:ee Choonsik, Kim KP, et al. radiation exposure from CT scans in childhood and subsequent risk of leukaemia and brain tumours: a retrospective cohort study. Lancet. 2012;380:499–505.CrossRef Pearce MS, Salotti JA, Little MP, McHugh K. L:ee Choonsik, Kim KP, et al. radiation exposure from CT scans in childhood and subsequent risk of leukaemia and brain tumours: a retrospective cohort study. Lancet. 2012;380:499–505.CrossRef
24.
go back to reference Zwaenepoel T, Vlam K. Sapho: treatment options including bisphosphonates. Semin Arthritis Rheum. 2016;46:168–73.CrossRef Zwaenepoel T, Vlam K. Sapho: treatment options including bisphosphonates. Semin Arthritis Rheum. 2016;46:168–73.CrossRef
Metadata
Title
Chronic non-bacterial osteomyelitis: a comparative study between children and adults
Authors
Andrea Skrabl-Baumgartner
Peter Singer
Theresa Greimel
Gregor Gorkiewicz
Josef Hermann
Publication date
01-12-2019
Publisher
BioMed Central
Published in
Pediatric Rheumatology / Issue 1/2019
Electronic ISSN: 1546-0096
DOI
https://doi.org/10.1186/s12969-019-0353-2

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