CASE REPORT
Maxillofacial Surgery
doi: 10.25005/2074-0581-2025-27-2-514-524
PRIMARY CHRONIC OSTEOMYELITIS OF THE JAW IN CHILDREN: TWO CASE REPORTS

A.YU. KUGUSHEV1, A.B. LOPATIN2, S. DAGHER2

1Department of Oral and Maxillofacial Surgery, Russian Childrens’ Clinical Hospital of the Pirogov Russian National Research Medical University, Moscow, Russian Federation
2Department of Oral and Maxillofacial Surgery, Peoples' Friendship University of Russia named after Patrice Lumumba, Moscow, Russian Federation

Methods: Between 2015 and 2023, forty-five children were treated for PCO at the Department of Oral and Maxillofacial Surgery, Russian Childrens’ Clinical Hospital, affiliated with the Pirogov Russian National Research Medical University. Of these, 17 children received combination therapy involving resection with removal of all granulomatous tissue, followed by postoperative administration of denosumab. From this group, two patients – a 15-yearold boy and a 6-year-old girl – were selected for this article due to their differing anthropometric characteristics and recurrent disease course.

Results: The treatment applied in these two clinical cases effectively increased the density of the affected bone area. However, if small cystic elements were not fully removed and debridement of granulated tissues was insufficient, relapse occurred, necessitating further surgical intervention. Postoperative use of denosumab and ibandronate contributed to maintaining disease remission.

Conclusion: It is crucial to determine the appropriate dosage, treatment duration, and drug administration frequency to achieve optimal outcomes and mitigate potential postoperative complications typically associated with the prescribed medications.

Keywords: Primary chronic osteomyelitis, rare diseases, autoinflammatory bone diseases, denosumab, antiresorptive therapy, bisphosphonates, case report.

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References
  1. Brøns C, Buch K, Schwarz P, Thuesen AC. Chronic non-bacterial osteomyelitis: A review. Calcified Tissue International. 2019;104(5):544-53. https://doi. org/10.1007/s00223-018-0495-0
  2. Kaiser D, Bolt I. Hofer M, Relly C, Berthet G, Bolz D, et al. Chronic nonbacterial osteomyelitis in children: A retrospective multicenter study. Pediatric Rheumatology. 201;13(25):1-7.‏ https://doi.org/10.1186/s12969-015-0023-y
  3. Cimaz R, Pastore S, Taddio A, Zennaro F. An update on the pathogenesis and treatment of chronic recurrent multifocal osteomyelitis in children. Pediatric Drugs. 2017;19(12):165-72. https://doi.org/10.1007/s40272-017-0226-4
  4. Kim SM, Lee SK. Chronic non-bacterial osteomyelitis in the jaw. Journal of the Korean Association of Oral and Maxillofacial Surgeons. 2019;45(2):68-75. https:// doi.org/10.5125/jkaoms.2019.45.2.68
  5. Kraus R, Laxer RM. Characteristics, treatment options, and outcomes of chronic non-bacterial osteomyelitis in children. Current Treatment Options in Rheumatology. 2020;6:205-22.‏ https://doi.org/10.5125/10.1007/s40674-020-00149-8
  6. Costa-Reis P, Sullivan KE. Chronic recurrent multifocal osteomyelitis. Journal of Clinical Immunology. 2013;33(6):1043-56. https://doi.org/10.1007/s10875-013- 9902-5
  7. Ferrara G, Finetti M, Insalaco A, Pardeo M, Gattorno M, Gregori M, et al. Dealing with chronic non-bacterial osteomyelitis: A practical approach. Pediatr Rheumatol Online J. 2017;15(1):87-95. https://doi.org/10.1186/s12969-017-0216-7
  8. Burgner D, Manners PJ, Murray KJ, Vercoe J, Walsh P. Chronic recurrent multifocal osteomyelitis in children: Nine years’ experience at a statewide tertiary paediatric rheumatology referral centre. Rheumatology. 2015;54(9):1688-91. https://doi. org/10.1093/rheumatology/kev013
  9. Eisenstein EM, Syverson GD, Vora SS, Williams CB. Combination therapy with methotrexate and etanercept for refractory chronic recurrent multifocal osteomyelitis. J Rheumatol. 2011;38(4):782-3. https://doi.org/10.3899/ jrheum.100431
  10. van de Meent MM, Appelman-Dijkstra NM, Wetselaar-Glas MJ, Pichardo SE, van Merkesteyn JR. Bisphosphonate therapy in chronic diffuse sclerosing osteomyelitis/tendoperiostitis of the mandible: Retrospective case series. Journal of Cranio-Maxillofacial Surgery. 2022;50(7):599-604. https://doi. org/10.1016/j.jcms.2022.05.011
  11. Finn A, Ramanan AV, Roderick M, Shah R. Efficacy of pamidronate therapy in children with chronic non-bacterial osteitis: Disease activity assessment by whole body magnetic resonance imaging. Rheumatology (Oxford). 2014;53(11):1973-6. https://doi.org/10.1093/rheumatology/keu226
  12. Burian E, Ehrenfeld M, Kaeppler G, Otto S, Troeltzsch M. Denosumab as a potential treatment alternative for patients suffering from diffuse sclerosing osteomyelitis of the mandible – A rapid communication. Journal of Cranio-Maxillofacial Surgery. 2018;46(4):534-7. https://doi.org/10.1016/j.jcms.2017.10.011
  13. Hallmer F, Bjornland T, Andersson G, Becktor JP, Kristoffersen AK, Enersen M. Bacterial diversity in medication‐related osteonecrosis of the jaw. Oral Surg Oral Med Oral Pathol Oral Radiol. 2017;123(4):436-44. https://doi.org/10.1016/j. oooo.2016.11.011
  14. Campbell TN, Frizzell B, MacMullan P. Chronic non-bacterial osteomyelitis masquerading as fibrous dysplasia. Modern Rheumatology Case Reports. 2020;4(2):192-5. https://doi.org/10.1080/24725625.2020.1728062
  15. Burian E, Ehrenfeld M, Mahaini S, Pautke C, Probst F, Smolka W, et al. Ibandronate treatment of diffuse sclerosing osteomyelitis of the mandible: Pain relief and insight into pathogenesis. Journal of Cranio-Maxillofacial Surgery.2015;43(9):1837-42. https://doi.org/10.1016/j.jcms.2015.08.02
  16. Bjørnland T, Hallmer F, Korduner M, Møystad A. Treatment of diffuse sclerosing osteomyelitis of the jaw with denosumab shows remarkable results – A report of two cases. Clinical Case Reports. 2018;6(12):2434-7. https://doi.org/10.1002/ ccr3.1894
  17. Kugushev AYu, Lopatin AV, Yasonov SA, Grachyov NS. Opyt ispol’zovaniya targetnoy terapii pri diffuzno-skleroziruyushchem osteomielite chelyustey u detey [Experience with the targeted treatment of diffuse sclerosing jaw osteomyelitis in children]. Golova i sheya. 2020;8(3):44-53. https://doi. org/10.25792/HN.2020.8.3.44–53
  18. Andersson G, Becktor JP, Bjørnland T, Hallmer F, Nicklasson A. Osteonecrosis of the jaw in patients treated with oral and intravenous bisphosphonates: Experience in Sweden. Oral Surg Oral Med Oral Pathol Oral Radiol. 2014;118(2):202-8. https:// doi.org/10.1016/j.oooo.2014.05.006

Authors' information:

Kugushev Aleksandr Yurievich,
Doctor of Medical Sciences, Maxillofacial Surgeon, Department of Oral and Maxillofacial Surgery, Russian Children's Clinical Hospital of the Pirogov Russian National Research Medical University
ORCID ID: 0000-0002-6881-7709
E-mail: drkuguzhev@gmail.com

Lopatin Andrey Vyachslavovich,
Doctor of Medical Sciences, Full Professor, Professor of the Department of Oral and Maxillofacial Surgery, Peoples' Friendship University of Russia named after Patrice Lumumba
ORCID ID: 0000-0003-0043-9059
E-mail: and-lopatin@yandex.ru

Dagher Suzan,
PhD Student of the Department of Oral and Maxillofacial Surgery, Peoples' Friendship University of Russia named after Patrice Lumumba
ORCID ID: 0009-0004-1950-4011
E-mail: suzandager@yandex.com

Information about support in the form of grants, equipment, medications

The authors did not receive financial support from manufacturers of medicines and medical equipment

Conflicts of interest: No conflict

Address for correspondence:

Dagher Suzan
PhD Student of the Department of Oral and Maxillofacial Surgery, Peoples' Friendship University of Russia named after Patrice Lumumba

117198, Russian Federation, Moscow, Miklukho-Maklay str., 10

Tel.: +7 (991) 6114298

E-mail: suzandager@yandex.com


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