Conservative treatment of chronic nonbacterial osteomyelitis using zoledronic acid in children
- 作者: Tairov G.N.1, Nazarenko A.G.1, Ochkurenko A.A.1, Kuleshov A.A.1, Vetrile M.S.1, Lisyansky I.N.1, Makarov S.N.1, Strunina U.V.2
-
隶属关系:
- Priorov National Medical Research Centre for Traumatology and Orthopaedics
- Burdenko National Medical Research Centre for Neurosurgery
- 期: 卷 32, 编号 2 (2025)
- 页面: 413-423
- 栏目: Original study articles
- URL: https://ogarev-online.ru/0869-8678/article/view/314748
- DOI: https://doi.org/10.17816/vto637078
- EDN: https://elibrary.ru/ACDZRS
- ID: 314748
如何引用文章
详细
BACKGROUND: Currently, there is no etiological treatment for chronic nonbacterial osteomyelitis. The insufficient efficacy of all available treatment modalities remains a major concern. Among the most effective approaches are genetically engineered therapy and bisphosphonate treatment. Pamidronate is the most frequently reported option in scientific publications. However, given pamidronates’ lower efficacy compared to zoledronic acid, we developed a treatment protocol that includes zoledronic acid at a dose of 0.05 mg/kg every 3 months, three infusions in total, along with active vitamin D metabolites and calcium carbonate.
AIM: This study aimed to demonstrate the efficacy of zoledronic acid in the treatment of chronic nonbacterial osteomyelitis.
METHODS: The study included 22 children aged 6 to 17 years. A prospective pilot study was conducted to assess the efficacy of zoledronic acid in children with chronic recurrent multifocal osteomyelitis. All patients underwent biopsy with morphological and microbiological verification of the diagnosis, as well as laboratory and imaging assessments before and 3, 6, and 12 months after treatment. Clinical disease activity was assessed using a visual analog scale for pain and the PedsQL 4.0 quality of life questionnaire.
RESULTS: Preliminary treatment outcomes in patients receiving this regimen are promising. Pain was significantly reduced, quality of life improved, and the number of bone lesions decreased, with clinical remission achieved in all patients.
CONCLUSION: Zoledronic acid rapidly inhibits osteoclast activity, leading to both clinical and radiological remission, as evidenced by decreased pain, reduction of bone marrow edema on MRI, and sclerosis of lytic lesions. Given the reduced osteoclast activity in the post-injection period, this therapy must be combined with active vitamin D metabolites and calcium carbonate to maintain calcium-phosphorus homeostasis.
作者简介
Gazinur Tairov
Priorov National Medical Research Centre for Traumatology and Orthopaedics
编辑信件的主要联系方式.
Email: gazinur.vezunchik@mail.ru
ORCID iD: 0009-0002-3469-3944
SPIN 代码: 8868-2577
MD
俄罗斯联邦, 10 Priorova st, Moscow, 127299Anton Nazarenko
Priorov National Medical Research Centre for Traumatology and Orthopaedics
Email: nazarenkoag@cito-priorov.ru
ORCID iD: 0000-0003-1314-2887
SPIN 代码: 1402-5186
Corresponding Member of the Russian Academy of Sciences, MD, Dr. Sci. (Medicine), Professor of RAS
俄罗斯联邦, 10 Priorova st, Moscow, 127299Alexander Ochkurenko
Priorov National Medical Research Centre for Traumatology and Orthopaedics
Email: cito-omo@mail.ru
ORCID iD: 0000-0002-1078-9725
SPIN 代码: 8324-2383
MD, Dr. Sci. (Medicine), Professor
俄罗斯联邦, 10 Priorova st, Moscow, 127299Alexander Kuleshov
Priorov National Medical Research Centre for Traumatology and Orthopaedics
Email: cito-spine@mail.ru
ORCID iD: 0000-0002-9526-8274
SPIN 代码: 7052-0220
MD, Dr. Sci. (Medicine)
俄罗斯联邦, 10 Priorova st, Moscow, 127299Marchel Vetrile
Priorov National Medical Research Centre for Traumatology and Orthopaedics
Email: vetrilams@cito-priorov.ru
ORCID iD: 0000-0001-6689-5220
SPIN 代码: 9690-5117
MD, Cand. Sci. (Medicine)
俄罗斯联邦, 10 Priorova st, Moscow, 127299Igor Lisyansky
Priorov National Medical Research Centre for Traumatology and Orthopaedics
Email: lisigornik@list.ru
ORCID iD: 0000-0002-2479-4381
SPIN 代码: 9845-1251
MD, Cand. Sci. (Medicine)
俄罗斯联邦, 10 Priorova st, Moscow, 127299Sergey Makarov
Priorov National Medical Research Centre for Traumatology and Orthopaedics
Email: moscow.makarov@gmail.com
ORCID iD: 0000-0003-0406-1997
SPIN 代码: 2767-2429
MD, Cand. Sci. (Medicine)
俄罗斯联邦, 10 Priorova st, Moscow, 127299Uliya Strunina
Burdenko National Medical Research Centre for Neurosurgery
Email: ustrunina@nsi.ru
ORCID iD: 0000-0001-5010-6661
SPIN 代码: 9799-5066
MD
俄罗斯联邦, Moscow参考
- Zhao DY, McCann L, Hahn G, Hedrich CM. Chronic nonbacterial osteomyelitis (CNO) and chronic recurrent multifocal osteomyelitis (CRMO). J Transl Autoimmun. 2021;4:100095. doi: 10.1016/j.jtauto.2021.100095
- Young S, Sharma N, Lee JH, et al. Mast cells enhance sterile inflammation in chronic nonbacterial osteomyelitis. Dis Model Mech. 2019;12(8):dmm040097. doi: 10.1242/dmm.040097
- Hofmann SR, Kapplusch F, Mäbert K, Hedrich CM. The molecular pathophysiology of chronic non-bacterial osteomyelitis (CNO) — a systematic review. Mol Cell Pediatr. 2017;4(1):7. doi: 10.1186/s40348-017-0073-y
- Hedrich CM, Morbach H, Reiser C, Girschick HJ. New Insights into Adult and Paediatric Chronic Non-bacterial Osteomyelitis CNO. Curr Rheumatol Rep. 2020;22(9):1–11. doi: 10.1007/s11926-020-00928-1
- Kostik MM, Kopchak OL, Chikova IA, et al. Differentiated approach to non-bacterial osteomyelitis treatment in children: The retrospective study results. Vopr Sovrem Pediatr. 2016;15(5):505–512. doi: 10.15690/vsp.v15i5.1625 EDN: WZKNDB
- Wipff J, Costantino F, Lemelle I, et al. A large national cohort of French patients with chronic recurrent multifocal osteitis. Arthritis Rheumatol. 2015;67(4):1128–37. doi: 10.1002/art.39013
- Borzutzky A, Stern S, Reiff A, et al. Pediatric chronic nonbacterial osteomyelitis. Pediatrics. 2012;130(5):e1190–7. doi: 10.1542/peds.2011-3788
- 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):1058–1065. doi: 10.3899/jrheum.161255
- Girschick H, Finetti M, Orlando F, 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. doi: 10.1093/rheumatology/key058
- Kostik MM, Kopchak OL, Chikova IA, Isupova EA, Mushkin AY. Comparison of different treatment approaches of pediatric chronic non-bacterial osteomyelitis. Rheumatol Int. 2019;39(1):89–96. doi: 10.1007/s00296-018-4151-9
- Just A, Adams S, Brinkmeier T, et al. Successful treatment of primary chronic osteomyelitis in SAPHO syndrome with bisphosphonates. J Dtsch Dermatol Ges. 2008;6(8):657–60. (in German). doi: 10.1111/j.1610-0387.2008.06588.x
- Tairov GN, Toptygina AP, Ochkurenko AA, Buklemishev YV, Karpov IN. Experience of successful treatment of a patient with chronic non-bacterial osteomyelitis (clinical case). N.N. Priorov J Traumatol Orthop. 2022;29(4):335–343. doi: 10.17816/vto111823 EDN: CCJWJO
- Kellinsalmi M, Mönkkönen H, Mönkkönen J, et al. In vitro comparison of clodronate, pamidronate and zoledronic acid effects on rat osteoclasts and human stem cell-derived osteoblasts. Basic Clin Pharmacol Toxicol. 2005;97(6):382–91. doi: 10.1111/j.1742-7843.2005.pto_176.x
- Jansson A, Renner ED, Ramser J, 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. doi: 10.1093/rheumatology/kel190
- Belova NА, Kostik MM, Buklaev DS, et al. Federal’nye klinicheskie rekomendatsii (protokol) po okazaniyu meditsinskoj pomoshhi patsientam s nesovershennym osteogenezom. Moscow; 2015. Available at: http://мороздгкб.рф/wp-content/uploads/2017/03/Федеральные-клинические-рекомендации-протокол-по-оказанию-медицинской-помощи-пациентам-с-несовершенным-остеогенезом.pdf. (In Russ.).
- Li C, Zhao Y, Zuo Y, et al. Efficacy of bisphosphonates in patients with synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: a prospective open study. Clin Exp Rheumatol. 2019;37(4):663–669.
- 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(9):1105–11. doi: 10.1007/s00431-010-1188-5
- Khanna G, Sato TSP, Ferguson P. Imaging of chronic recurrent Multifocal Osteomyelitis. Radiographics. 2009;29(4):1159–77. doi: 10.1148/rg.294085244
- Zhao Y, Wu EY, Oliver MS, et al. Consensus Treatment Plans for Chronic Nonbacterial Osteomyelitis Refractory to Nonsteroidal Antiinflammatory Drugs and/or With Active Spinal Lesions. Arthritis Care Res (Hoboken). 2018;70(8):1228–1237. doi: 10.1002/acr.23462
- Petukhova V, Mushkin A, Kostik M. Bisphosphonate treatment in the bone disorders in children: a systematic review. MedAlliance. 2021;9(3):59–70. doi: 10.36422/23076348-2021-9-3-59-70 EDN: JSMWWT
补充文件
