Clinical Characteristics and Short-term Outcomes of Paediatric Patients with Chronic Recurrent Multifocal Osteomyelitis
Year 2024,
, 334 - 338, 20.02.2024
Özge Baba
,
Hakan Kısaoğlu
,
Mukaddes Kalyoncu
Abstract
Objective: Chronic recurrent multifocal osteomyelitis (CRMO) is the most common autoinflammatory disease of the bone characterized by pain and inflammatory lesions without an infectious agent. The aim of this study is to evaluate the clinical, laboratory, and imaging features and treatments of paediatric patients with CRMO followed in our pediatric rheumatology clinic.
Material and Methods: Medical records of ten patients diagnosed with CRMO according to Bristol diagnostic criteria between January 2018 and June 2021 were retrospectively reviewed.
Results: The mean age at diagnosis was 9.3 years, and eight of the patients were male. One patient had concomitant psoriasis, two patients had familial Mediterranean fever and one patient had a history of immunoglobulin A vasculitis. Half of the patients had a moderate acute phase reactant elevation. The most frequently involved bones were the lower extremity bones. While localized magnetic resonance imaging (MRI) was the most commonly used imaging modality in the diagnosis of CRMO, silent bone lesions (15%) were detected only by the whole-body MRI. Non-steroidal anti-inflammatory drug (NSAID) was given to all patients. As second-line therapy, methotrexate and pamidronate were employed on seven and five patients respectively. Three of the patients received anti-TNF treatment (etanercept and infliximab) as the third-line therapy. Imaging with whole-body MRI is important due to clinically silent bone lesions, especially in the course of CRMO.
Conclusion: Concomitant familial Mediterranean fever was not rare in cases with CRMO and treatment of CRMO might be challenging due to the need for anti-TNF treatment in a considerable number of patients.
References
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Year 2024,
, 334 - 338, 20.02.2024
Özge Baba
,
Hakan Kısaoğlu
,
Mukaddes Kalyoncu
References
- Hofmann SR, Kapplusch F, Girschick HJ, Morbach H, Pablik J, Ferguson PJ, et al. Chronic recurrent multifocal osteomyelitis (CRMO): presentation, pathogenesis, and treatment. Curr Osteoporos Rep 2017;15(6):542-54. google scholar
- Giedion A, Holthusen W, Masel LF, Vischer D. [Subacute and chronic “symmetrical” osteomyelitis]. Ann Radiol 1972;15(3):329-42. google scholar
- Björksten B, Gustavson KH, Eriksson B, Lindholm A, Nordström S. Chronic recurrent multifocal osteomyelitis and pustulosis palmoplantaris. J Pediatr 1978;93(2):227-31. google scholar
- Buch K, Thuesen ACB, Br0ns C, Schwarz P. Chronic non-bacterial osteomyelitis: a review. Calcif Tissue Int 2019;104(5):544-53. google scholar
- 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 2015;54(9):1688-91 google scholar
- Jansson AF, Grote V; ESPED Study Group. Nonbacterial osteitis in children: data of a German Incidence Surveillance Study. Acta Paediatr 2011;100(8):1150-7. google scholar
- Chen Z, Cheng L, Feng G. Bone inflammation and chronic recurrent multifocal osteomyelitis. Eur Rev Med Pharmacol Sci 2018; 22(5): 1380-6. google scholar
- Roderick MR, Shah R, Rogers V, Finn A, Ramanan AV. Chronic recurrent multifocal osteomyelitis (CRMO) - advancing the diagnosis. Pediatr Rheumatol Online J 2016;14(1):47. google scholar
- 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 2018;57(8):1203-11. google scholar
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- 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(7):1058-65. google scholar
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- Watad A, Bragazzi NL, Adawi M, Shoenfeld Y, Comaneshter D, Cohen AD, et al. FMF is associated with a wide spectrum of MHC class I- and allied SpA disorders but not with classical MHC class II-asssociated autoimmune disease: insights from a large cohort study. Front Immunol 2019;10:2733. google scholar
- 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. google scholar
- Ozer E, Seker D, Taner E, Adrovic A, Sahin S, Barut K, et al. The frequency of juvenile spondyloarthropathies in childhood familial Mediterranean fever. Clin Exp Rheumatol 2018;36(6 Suppl 115):141-5. google scholar
- Sönmez HE, Batu ED, Demir S, Bilginer Y, Özen S. Comparison of patients with familial Mediterranean fever accompanied with sacroiliitis and patients with juvenile spondyloarthropathy. Clin Exp Rheumatol 2017;108(6):124-7. google scholar
- Zhao DY, McCann L, Hahn G, Hedrich CM. Chronic nonbacterial osteomyelitis (CNO) and chronic recurrent multifocal osteomyelitis (CRMO). J Transl Autoimmun 2021;4:100095. google scholar
- 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 2008;47(9):1397-9. google scholar
- Sözeri B, Ayaz NA, Atıkan BY, Karadağ ŞG, Çakan M, Argın M, et al. Clinical experiences in Turkish paediatric patients with chronic recurrent multifocal osteomyelitis. Turk J Pediatr 2019;61(6):879-84. google scholar
- Sato TS, Watal P, Ferguson PJ. Imaging mimics of chronic recurrent multifocal osteomyelitis: avoiding pitfalls in a diagnosis of exclusion. Pediatr Radiol 2020;50(1):124-36. google scholar
- 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(4):578-82. google scholar
- Juszczak B, Sutko J. Patient-reported effectiveness and safety of Pamidronate in NSAIDs refractory chronic recurrent multifocal osteomyelitis in children. Rheumatol Int 2022;42(4):699-706. google scholar
- 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(4):707-12. google scholar
- Simm PJ, Allen RC, Zacharin MR. Bisphosphonate treatment in chronic recurrent multifocal osteomyelitis. J Pediatr 2008;152(4):571-5. google scholar
- Batu ED, Ergen FB, Gulhan B, Topaloglu R, Aydingoz U, Ozen S. Etanercept treatment in five cases of refractory chronic recurrent multifocal osteomyelitis (CRMO). Joint Bone Spine 2015;82(6):471-3. google scholar
- Zhao Y, Wu EY, Oliver MS, Cooper AM, Basiaga ML, Vora SS, et al. Consensus treatment plans for chronic nonbacterial osteomyelitis refractory to nonsteroidal antiinflammatory drugs and/or with active spinal lesions. Arthritis Care Res 2018;70(8):1228-37. google scholar