Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis

Yıl: 2023 Cilt: 53 Sayı: 5 Sayfa Aralığı: 1105 - 1111 Metin Dili: İngilizce DOI: 10.55730/1300-0144.5675 İndeks Tarihi: 24-11-2023

Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis

Öz:
Background/aim: Chronic nonbacterial osteomyelitis (CNO) is a rare disease of unknown etiology and most commonly occurs during childhood or adolescence. The purpose of this study is to collect data on the clinical features, outcomes, and management of the disease and to identify the factors affecting recurrence. Materials and methods: This is a retrospective multicenter cross-sectional study of pediatric patients diagnosed with CNO. A total of 87 patients with a diagnosis of CNO followed for at least 6 months in 8 pediatric rheumatology centers across the country between January 2010 and December 2021 were included in this study. Results: The study included 87 patients (38 girls, 49 boys; median age: 12.5 years). The median follow-up time was 20 months (IQR: 8.5–40). The median time of diagnostic delay was 9.9 months (IQR: 3–24). Arthralgia and bone pain were the most common presenting symptoms. Multifocal involvement was detected in 86.2% of the cases and a recurrent course was reported in one-third of those included in the study. The most commonly involved bones were the femur and tibia. Vertebrae and clavicles were affected in 19.5% and 20.6% of cases, respectively. The erythrocyte sedimentation rate (ESR) values of 60.9% of the patients were above 20 mm/h and the C-reactive protein values of 44.8% were above 5 mg/L. The remission rate was 13.3% in patients using nonsteroidal antiinflammatory drugs and 75.0% in those using biological drugs. Vertebral and mandibular involvement and high ESR values at the time of diagnosis were associated with recurrence. Conclusion: In this multicenter study, CNO with vertebral and mandibular involvement and high ESR at diagnosis were associated with recurrence.
Anahtar Kelime: Chronic nonbacterial osteomyelitis chronic recurrent multifocal osteomyelitis autoinflammation children

Belge Türü: Makale Makale Türü: Araştırma Makalesi Erişim Türü: Erişime Açık
  • 1. Girschick H, Finetti M, Orlando F, Schalm, S Insalaco A et al. The multifaceted presentation of chronic recurrent multifocal osteomyelitis: a series of 486 cases from the Eurofever international registry. Rheumatology (Oxford) 2018; 57 (7): 1203-1211. https:// doi.org/10.1093/rheumatology/key058
  • 2. Nuruzzaman F, Zhao Y, Ferguson PJ. Chronic nonbacterial osteomyelitis: insights into pathogenesis, assessment, and treatment. Rheumatic Diseases Clinics of North America 2021; 47 (4): 691-705. https://doi.org/10.1016/j.rdc.2021.06.005
  • 3. Zhao Y, Wu EY, Oliver MS, Cooper AM, Basiaga ML et al. Consensus treatment plans for chronic nonbacterial osteomyelitis refractory to nonsteroidal antiinflammatory drugs and/or with active spinal lesions. Arthritis Care & Research 2018; 70 (8): 1228- 1237. https://doi.org/10.1002/acr.23462
  • 4. Hedrich CM, Morbach H, Reiser C, Girschick HJ. New insights into adult and paediatric chronic non-bacterial osteomyelitis CNO. Current Rheumatology Reports 2020; 22 (9): 52. https:// doi.org/10.1007/s11926-020-00928-1
  • 5. Ramanan AV, Hampson LV, Lythgoe H, Jones AP, Hardwick B et al. Defining consensus opinion to develop randomised controlled trials in rare diseases using Bayesian design: an example of a proposed trial of adalimumab versus pamidronate for children with CNO/CRMO. PLoS One 2019; 14 (6): e0215739. https://doi. org/10.1371/journal.pone.0215739
  • 6. Zhao DY, McCann L, Hahn G, Hedrich CM. Chronic nonbacterial osteomyelitis (CNO) and chronic recurrent multifocal osteomyelitis (CRMO). Journal of Translational Autoimmunity 2021; 4: 100095. https://doi.org/10.1016/j.jtauto.2021.100095
  • 7. SchnabelA,RangeU,HahnG,BernerR,HedrichCM.Treatment response and longterm outcomes in children with chronic nonbacterial osteomyelitis. Journal of Rheumatology 2017; 44 (7): 1058-1065. https://doi.org/10.3899/jrheum.161255
  • 8. Borzutzky A, Stern S, Reiff A, Steinberg EA, Dedeoglu F et al. Pediatric chronic nonbacterial osteomyelitis. Pediatrics 2012; 130 (5): e1190-e1197. https://doi.org/10.1542/peds.2011-3788
  • 9. Roderick MR, Shah R, Rogers V, Finn A, Ramanan AV. Chronic recurrent multifocal osteomyelitis (CRMO) - advancing the diagnosis. Pediatric Rheumatology Online Journal 2016; 14 (1): 47. https://doi.org/10.1186/s12969-016-0109-1
  • 10. d’Angelo P, de Horatio LT, Toma P, Ording Müller LS, Avenarius D et al. Chronic nonbacterial osteomyelitis - clinical and magnetic resonance imaging features. Pediatric Radiology 2021; 51 (2): 282-288. https://doi.org/10.1007/s00247-020- 04827-6
  • 11. Wipff J, Costantino F, Lemelle I, Pajot C, Duquesne A et al. A large national cohort of French patients with chronic recurrent multifocal osteitis. Arthritis & Rheumatology 2015; 67 (4): 1128-1137. https://doi.org/10.1002/art.39013
  • 12. Sözeri B, Ayaz NA, Atıkan BY, Karadağ ŞG, Çakan M et al. Clinical experiences in Turkish paediatric patients with chronic recurrent multifocal osteomyelitis. Turkish Journal of Pediatrics 2019; 61 (6): 879-884. https://doi.org/10.24953/ turkjped.2019.06.009
  • 13. Concha S, Hernández-Ojeda A, Contreras O, Mendez C, Talesnik E et al. Chronic nonbacterial osteomyelitis in children: a multicenter case series. Rheumatology International 2020; 40 (1): 115-120. https://doi.org/10.1007/s00296-019-04400-x
  • 14. Okay E, Ulu K, Demir F, Sari T, Zeynalov S et al. Chronic recurrent multifocal osteomyelitis: a multidisciplinary experience of 22 pediatric cases with a mean follow-up of 27 months. Journal of Orthopaedic Science 2023; 28 (2): 438-445. https://doi.org/10.1016/j.jos.2021.11.009
  • 15. Bhat CS, Anderson C, Harbinson A, McCann LJ, Roderick M et al. Chronic nonbacterial osteitis- a multicentre study. Pediatric Rheumatology Online Journal 2018; 16 (1): 74. https://doi.org/10.1186/s12969-018-0290-5
  • 16. Açarı C, Çomak E, Çekiç Ş, Türkuçar S, Adıgüzel Dündar H et al. Clinical features of children with chronic non-bacterial osteomyelitis: a multicenter retrospective case series from Turkey. Archives of Rheumatology 2021; 36 (3): 419-426. https://doi.org/10.46497/ArchRheumatol.2021
  • 17. Ma L, Liu H, Tang H, Zhang Z, Zou L et al. Clinical characteristics and outcomes of chronic nonbacterial osteomyelitis in children: a multicenter case series. Pediatric Rheumatology Online Journal 2022; 20 (1): 1. https://doi. org/10.1186/s12969-021-00657-4
  • 18. Kostik MM, Kopchak OL, Maletin AS, Mushkin AY. The peculiarities and treatment outcomes of the spinal form of chronic non-bacterial osteomyelitis in children: a retrospective cohort study. Rheumatology International 2020; 40 (1): 97-105. https://doi.org/10.1007/s00296-019-04479-2
  • 19. Gaal A, Basiaga ML, Zhao Y, Egbert M. Pediatric chronic nonbacterial osteomyelitis of the mandible: Seattle Children’s Hospital 22-patient experience. Pediatric Rheumatology Online Journal 2020; 18 (1): 4. https://doi.org/10.1186/s12969- 019-0384-8
  • 20. Schnabel A, Nashawi M, Anderson C, Felsenstein S, Lamoudi M et al. TNF-inhibitors or bisphosphonates in chronic nonbacterial osteomyelitis? - Results of an international retrospective multicenter study. Clinical Immunology 2022; 238: 109018. https://doi.org/10.1016/j.clim.2022.109018
  • 21. Timme M, Bohner L, Huss S, Kleinheinz J, Hanisch M. Response of different treatment protocols to treat chronic non- bacterial osteomyelitis (CNO) of the mandible in adult patients: a systematic review. International Journal of Environmental Research and Public Health 2020; 17 (5): 1737. https://doi. org/10.3390/ijerph17051737
  • 22. Andreasen CM, Jurik AG, Glerup MB, Høst C, Mahler BT et al. Response to early-onset pamidronate treatment in chronic nonbacterial osteomyelitis: a retrospective single-center study. Journal of Rheumatology 2019; 46 (11): 1515-1523. https://doi.org/10.3899/jrheum.181254
APA ULU K, İşgüder R, Karadağ Ş, Bağlan E, Kavrul Kayaalp G, otar yener g, Ozturk K, Sönmez H, Özdel S, demir f, Makay B, Unsal E, sozeri b, Aktay Ayaz N, Çakan M (2023). Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis. , 1105 - 1111. 10.55730/1300-0144.5675
Chicago ULU KADİR,İşgüder Rana,Karadağ Şerife Gül,Bağlan Esra,Kavrul Kayaalp Gülşah,otar yener gulcin,Ozturk Kubra,Sönmez Hafize Emine,Özdel Semanur,demir ferhat,Makay Balahan,Unsal Erbil,sozeri betul,Aktay Ayaz Nuray,Çakan Mustafa Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis. (2023): 1105 - 1111. 10.55730/1300-0144.5675
MLA ULU KADİR,İşgüder Rana,Karadağ Şerife Gül,Bağlan Esra,Kavrul Kayaalp Gülşah,otar yener gulcin,Ozturk Kubra,Sönmez Hafize Emine,Özdel Semanur,demir ferhat,Makay Balahan,Unsal Erbil,sozeri betul,Aktay Ayaz Nuray,Çakan Mustafa Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis. , 2023, ss.1105 - 1111. 10.55730/1300-0144.5675
AMA ULU K,İşgüder R,Karadağ Ş,Bağlan E,Kavrul Kayaalp G,otar yener g,Ozturk K,Sönmez H,Özdel S,demir f,Makay B,Unsal E,sozeri b,Aktay Ayaz N,Çakan M Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis. . 2023; 1105 - 1111. 10.55730/1300-0144.5675
Vancouver ULU K,İşgüder R,Karadağ Ş,Bağlan E,Kavrul Kayaalp G,otar yener g,Ozturk K,Sönmez H,Özdel S,demir f,Makay B,Unsal E,sozeri b,Aktay Ayaz N,Çakan M Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis. . 2023; 1105 - 1111. 10.55730/1300-0144.5675
IEEE ULU K,İşgüder R,Karadağ Ş,Bağlan E,Kavrul Kayaalp G,otar yener g,Ozturk K,Sönmez H,Özdel S,demir f,Makay B,Unsal E,sozeri b,Aktay Ayaz N,Çakan M "Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis." , ss.1105 - 1111, 2023. 10.55730/1300-0144.5675
ISNAD ULU, KADİR vd. "Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis". (2023), 1105-1111. https://doi.org/10.55730/1300-0144.5675
APA ULU K, İşgüder R, Karadağ Ş, Bağlan E, Kavrul Kayaalp G, otar yener g, Ozturk K, Sönmez H, Özdel S, demir f, Makay B, Unsal E, sozeri b, Aktay Ayaz N, Çakan M (2023). Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis. Turkish Journal of Medical Sciences, 53(5), 1105 - 1111. 10.55730/1300-0144.5675
Chicago ULU KADİR,İşgüder Rana,Karadağ Şerife Gül,Bağlan Esra,Kavrul Kayaalp Gülşah,otar yener gulcin,Ozturk Kubra,Sönmez Hafize Emine,Özdel Semanur,demir ferhat,Makay Balahan,Unsal Erbil,sozeri betul,Aktay Ayaz Nuray,Çakan Mustafa Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis. Turkish Journal of Medical Sciences 53, no.5 (2023): 1105 - 1111. 10.55730/1300-0144.5675
MLA ULU KADİR,İşgüder Rana,Karadağ Şerife Gül,Bağlan Esra,Kavrul Kayaalp Gülşah,otar yener gulcin,Ozturk Kubra,Sönmez Hafize Emine,Özdel Semanur,demir ferhat,Makay Balahan,Unsal Erbil,sozeri betul,Aktay Ayaz Nuray,Çakan Mustafa Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis. Turkish Journal of Medical Sciences, vol.53, no.5, 2023, ss.1105 - 1111. 10.55730/1300-0144.5675
AMA ULU K,İşgüder R,Karadağ Ş,Bağlan E,Kavrul Kayaalp G,otar yener g,Ozturk K,Sönmez H,Özdel S,demir f,Makay B,Unsal E,sozeri b,Aktay Ayaz N,Çakan M Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis. Turkish Journal of Medical Sciences. 2023; 53(5): 1105 - 1111. 10.55730/1300-0144.5675
Vancouver ULU K,İşgüder R,Karadağ Ş,Bağlan E,Kavrul Kayaalp G,otar yener g,Ozturk K,Sönmez H,Özdel S,demir f,Makay B,Unsal E,sozeri b,Aktay Ayaz N,Çakan M Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis. Turkish Journal of Medical Sciences. 2023; 53(5): 1105 - 1111. 10.55730/1300-0144.5675
IEEE ULU K,İşgüder R,Karadağ Ş,Bağlan E,Kavrul Kayaalp G,otar yener g,Ozturk K,Sönmez H,Özdel S,demir f,Makay B,Unsal E,sozeri b,Aktay Ayaz N,Çakan M "Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis." Turkish Journal of Medical Sciences, 53, ss.1105 - 1111, 2023. 10.55730/1300-0144.5675
ISNAD ULU, KADİR vd. "Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis". Turkish Journal of Medical Sciences 53/5 (2023), 1105-1111. https://doi.org/10.55730/1300-0144.5675