Journal of the Korean Association of Oral and Maxillofacial Surgeons

The Korean Association of Oral and Maxillofacial Surgeons (대한구강악안면외과학회)
권호 표지
DOI QR코드

DOI QR Code

Chronic non-bacterial osteomyelitis in the jaw

  • Kim, Soung Min (Oral and Maxillofacial Microvascular Reconstruction LAB, Brong Ahafo Regional Hospital) ;
  • Lee, Suk Keun (Department of Oral Pathology, College of Dentistry and Institute of Oral Science, Gangneung-Wonju National University)
  • Received : 2018.07.17
  • Accepted : 2018.08.21
  • Published : 2019.04.30
Download PDF
⟨ Previous Next ⟩

Abstract

Chronic recurrent multifocal osteomyelitis (CRMO) is one of the most severe form of chronic non-bacterial osteomyelitis (CNO), which could result in bone and related tissue damage. This autoinflammatory bone disorder (ABD) is very difficult for its clinical diagnosis because of no diagnostic criteria or biomarkers. CRMO in the jaw must be suspected in the differential diagnosis of chronic and recurrent bone pain in the jaw, and a bone biopsy should be considered in chronic and relapsing bone pain with swelling that is unresponsive to treatment. The early diagnosis of CRMO in the jaw will prevent unnecessary and prolonged antibiotic usage or unnecessary surgical intervention. The updated researches for the identification of genetic and molecular alterations in CNO/CRMO should be studied more for its correct pathophysiological causes and proper treatment guidelines. Although our trial consisted of reporting items from Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA), there are very few articles of randomized controlled trials. This article was summarized based on the author's diverse clinical experiences. This paper reviews the clinical presentation of CNO/CRMO with its own pathogenesis, epidemiology, recent research studies, and general medications. Treatment and monitoring of the jaw are essential for the clear diagnosis and management of CNO/CRMO patients in the field of dentistry and maxillofacial surgery.

Keywords

References

  1. 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. https://doi.org/10.3899/jrheum.160256
  2. 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;15:542-4. https://doi.org/10.1007/s11914-017-0405-9
  3. Giedion A, Holthusen W, Masel LF, Vischer D. Subacute and chronic "symmetrical" osteomyelitis. Ann Radiol (Paris) 1972;15:329-42.
  4. Andersson R. Effective treatment with interferon-alpha in chronic recurrent multifocal osteomyelitis. J Interferon Cytokine Res 1995;15:837-8. https://doi.org/10.1089/jir.1995.15.837
  5. Wang Y, Yang C, Zhang W, Lu Y, Wei W, Han Z, et al. Monofocal chronic nonbacterial osteomyelitis in the mandible accompanied with mucocutaneous disease. J Craniofac Surg 2017;28:e547-51. https://doi.org/10.1097/SCS.0000000000003840
  6. Padwa BL, Dentino K, Robson CD, Woo SB, Kurek K, Resnick CM. Pediatric chronic nonbacterial osteomyelitis of the jaw: clinical, radiographic, and histopathologic features. J Oral Maxillofac Surg 2016;74:2393-402. https://doi.org/10.1016/j.joms.2016.05.021
  7. Monsour PA, Dalton JB. Chronic recurrent multifocal osteomyelitis involving the mandible: case reports and review of the literature. Dentomaxillofac Radiol 2010;39:184-90. https://doi.org/10.1259/dmfr/23060413
  8. Hedrich CM, Hahn G, Girschick HJ, Morbach H. A clinical and pathomechanistic profile of chronic nonbacterial osteomyelitis/chronic recurrent multifocal osteomyelitis and challenges facing the field. Expert Rev Clin Immunol 2013;9:845-54. https://doi.org/10.1586/1744666X.2013.824670
  9. Hedrich CM, Hofmann SR, Pablik J, Morbach H, Girschick HJ. Autoinflammatory bone disorders with special focus on chronic recurrent multifocal osteomyelitis (CRMO). Pediatr Rheumatol Online J 2013;11:47. https://doi.org/10.1186/1546-0096-11-47
  10. Stern SM, Ferguson PJ. Autoinflammatory bone diseases. Rheum Dis Clin North Am 2013;39:735-49. https://doi.org/10.1016/j.rdc.2013.05.002
  11. El-Shanti HI, Ferguson PJ. Chronic recurrent multifocal osteomyelitis: a concise review and genetic update. Clin Orthop Relat Res 2007;462:11-9. https://doi.org/10.1097/BLO.0b013e3180986d73
  12. Borzutzky A, Stern S, Reiff A, Zurakowski D, Steinberg EA, Dedeoglu F, et al. Pediatric chronic nonbacterial osteomyelitis. Pediatrics 2012;130:e1190-7. https://doi.org/10.1542/peds.2011-3788
  13. Hofmann C, Wurm M, Schwarz T, Neubauer H, Beer M, Girschick H, et al. A standardized clinical and radiological follow-up of patients with chronic non-bacterial osteomyelitis treated with pamidronate. Clin Exp Rheumatol 2014;32:604-9.
  14. Khanna G, Sato TS, Ferguson P. Imaging of chronic recurrent multifocal osteomyelitis. Radiographics 2009;29:1159-77. https://doi.org/10.1148/rg.294085244
  15. Jurik AG, Egund N. MRI in chronic recurrent multifocal osteomyelitis. Skelet Radiol 1997;26:230-8. https://doi.org/10.1007/s002560050227
  16. 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.
  17. Beck C, Girschick HJ, Morbach H, Schwarz T, Yimam T, Frenkel J, et al. Mutation screening of the IL-1 receptor antagonist gene in chronic non-bacterial osteomyelitis of childhood and adolescence. Clin Exp Rheumatol 2011;29:1040-3.
  18. Golla A, Jansson A, Ramser J, Hellebrand H, Zahn R, Meitinger T, et al. Chronic recurrent multifocal osteomyelitis (CRMO): evidence for a susceptibility gene located on chromosome 18q21.3- 18q22. Eur J Hum Genet 2002;10:217-21. https://doi.org/10.1038/sj.ejhg.5200789
  19. Nakashima T, Takayanagi H. Osteoimmunology: crosstalk between the immune and bone systems. J Clin Immunol 2009;29:555-67. https://doi.org/10.1007/s10875-009-9316-6
  20. Nakashima T, Takayanagi H. Osteoclasts and the immune system. J Bone Miner Metab 2009;27:519-29. https://doi.org/10.1007/s00774-009-0089-z
  21. Sharma M, Ferguson PJ. Autoinflammatory bone disorders: update on immunologic abnormalities and clues about possible triggers. Curr Opin Rheumatol 2013;25:658-64. https://doi.org/10.1097/BOR.0b013e328363eb08
  22. Hamel J, Paul D, Gahr M, Hedrich CM. Pilot study: possible association of IL10 promoter polymorphisms with CRMO. Rheumatol Int 2012;32:555-6. https://doi.org/10.1007/s00296-010-1768-8
  23. Hofmann SR, Kubasch AS, Ioannidis C, Rosen-Wolff A, Girschick HJ, Morbach H, et al. Altered expression of IL-10 family cytokines in monocytes from CRMO patients result in enhanced IL-$1{\beta}$ expression and release. Clin Immunol 2015;161:300-7. https://doi.org/10.1016/j.clim.2015.09.013
  24. Hofmann SR, Morbach H, Schwarz T, Rosen-Wolff A, Girschick HJ, Hedrich CM. Attenuated TLR4/MAPK signaling in monocytes from patients with CRMO results in impaired IL-10 expression. Clin Immunol 2012;145:69-76. https://doi.org/10.1016/j.clim.2012.07.012
  25. Hofmann SR, Schwarz T, Moller JC, Morbach H, Schnabel A, Rosen-Wolff A, et al. Chronic non-bacterial osteomyelitis is associated with impaired Sp1 signaling, reduced IL10 promoter phosphorylation, and reduced myeloid IL-10 expression. Clin Immunol 2011;141:317-27. https://doi.org/10.1016/j.clim.2011.08.012
  26. Hedrich CM, Bream JH. Cell type-specific regulation of IL-10 expression in inflammation and disease. Immunol Res 2010;47:185-206. https://doi.org/10.1007/s12026-009-8150-5
  27. Hofmann SR, Rosen-Wolff A, Tsokos GC, Hedrich CM. Biological properties and regulation of IL-10 related cytokines and their contribution to autoimmune disease and tissue injury. Clin Immunol 2012;143:116-27. https://doi.org/10.1016/j.clim.2012.02.005
  28. Lindor NM, Arsenault TM, Solomon H, Seidman CE, McEvoy MT. A new autosomal dominant disorder of pyogenic sterile arthritis, pyoderma gangrenosum, and acne: PAPA syndrome. Mayo Clin Proc 1997;72:611-5. https://doi.org/10.1016/S0025-6196(11)63565-9
  29. Smith EJ, Allantaz F, Bennett L, Zhang D, Gao X, Wood G, et al. Clinical, molecular, and genetic characteristics of PAPA syndrome: a review. Curr Genomics 2010;11:519-27. https://doi.org/10.2174/138920210793175921
  30. Shreiner AB, Kao JY, Young VB. The gut microbiome in health and in disease. Curr Opin Gastroenterol 2015;31:69-75. https://doi.org/10.1097/MOG.0000000000000139
  31. Liao HJ, Chyuan IT, Wu CS, Lin SW, Chen KH, Tsai HF, et al. Increased neutrophil infiltration, IL-1 production and a SAPHO syndrome-like phenotype in PSTPIP2-deficient mice. Rheumatology (Oxford) 2015;54:1317-26. https://doi.org/10.1093/rheumatology/keu481
  32. Schnabel A, Range U, Hahn G, Siepmann T, Berner R, Hedrich CM. Unexpectedly high incidences of chronic non-bacterial as compared to bacterial osteomyelitis in children. Rheumatol Int 2016;36:1737-45. https://doi.org/10.1007/s00296-016-3572-6
  33. Morbach H, Hedrich CM, Beer M, Girschick HJ. Autoinflammatory bone disorders. Clin Immunol 2013;147:185-96. https://doi.org/10.1016/j.clim.2012.12.012
  34. Cassel SL, Janczy JR, Bing X, Wilson SP, Olivier AK, Otero JE, et al. Inflammasome-independent IL-$1{\beta}$ mediates autoinflammatory disease in Pstpip2-deficient mice. Proc Natl Acad Sci U S A 2014;111:1072-7. https://doi.org/10.1073/pnas.1318685111
  35. Chitu V, Nacu V, Charles JF, Henne WM, McMahon HT, Nandi S, et al. PSTPIP2 deficiency in mice causes osteopenia and increased differentiation of multipotent myeloid precursors into osteoclasts. Blood 2012;120:3126-35. https://doi.org/10.1182/blood-2012-04-425595
  36. Grosse J, Chitu V, Marquardt A, Hanke P, Schmittwolf C, Zeitlmann L, et al. Mutation of mouse Mayp/Pstpip2 causes a macrophage autoinflammatory disease. Blood 2006;107:3350-8. https://doi.org/10.1182/blood-2005-09-3556
  37. Greenhill CJ, Jones GW, Nowell MA, Newton Z, Harvey AK, Moideen AN, et al. Interleukin-10 regulates the inflammasomedriven augmentation of inflammatory arthritis and joint destruction. Arthritis Res Ther 2014;16:419. https://doi.org/10.1186/s13075-014-0419-y
  38. Scianaro R, Insalaco A, Bracci Laudiero L, De Vito R, Pezzullo M, Teti A, et al. Deregulation of the IL-$1{\beta}$ axis in chronic recurrent multifocal osteomyelitis. Pediatr Rheumatol Online J 2014;12:30. https://doi.org/10.1186/1546-0096-12-30
  39. Cox AJ, Darbro BW, Laxer RM, Velez G, Bing X, Finer AL, et al. Recessive coding and regulatory mutations in FBLIM1 underlie the pathogenesis of chronic recurrent multifocal osteomyelitis (CRMO). PLoS One 2017;12:e0169687. https://doi.org/10.1371/journal.pone.0169687
  40. Ferguson PJ, El-Shanti HI. Autoinflammatory bone disorders. Curr Opin Rheumatol 2007;19:492-8. https://doi.org/10.1097/BOR.0b013e32825f5492
  41. Jansson AF, Muller TH, Gliera L, Ankerst DP, Wintergerst U, Belohradsky BH, et al. Clinical score for nonbacterial osteitis in children and adults. Arthritis Rheum 2009;60:1152-9. https://doi.org/10.1002/art.24402
  42. Beck C, Morbach H, Beer M, Stenzel M, Tappe D, Gattenlohner S, et al. Chronic nonbacterial osteomyelitis in childhood: prospective follow-up during the first year of anti-inflammatory treatment. Arthritis Res Ther 2010;12:R74. https://doi.org/10.1186/ar2992
  43. Boehncke WH, Schon MP. Psoriasis. Lancet 2015;386:983-94. https://doi.org/10.1016/S0140-6736(14)61909-7
  44. Girschick HJ, Zimmer C, Klaus G, Darge K, Dick A, Morbach H. Chronic recurrent multifocal osteomyelitis: what is it and how should it be treated? Nat Clin Pract Rheumatol 2007;3:733-8. https://doi.org/10.1038/ncprheum0653
  45. Holden W, David J. Chronic recurrent multifocal osteomyelitis: two cases of sacral disease responsive to corticosteroids. Clin Infect Dis 2005;40:616-9. https://doi.org/10.1086/427217
  46. Miettunen PM, Wei X, Kaura D, Reslan WA, Aguirre AN, Kellner JD. Dramatic pain relief and resolution of bone inflammation following pamidronate in 9 pediatric patients with persistent chronic recurrent multifocal osteomyelitis (CRMO). Pediatr Rheumatol Online J 2009;7:2. https://doi.org/10.1186/1546-0096-7-2
  47. Compeyrot-Lacassagne S, Rosenberg AM, Babyn P, Laxer RM. Pamidronate treatment of chronic noninfectious inflammatory lesions of the mandible in children. J Rheumatol 2007;34:1585-9.
  48. Eleftheriou D, Gerschman T, Sebire N, Woo P, Pilkington CA, Brogan PA. Biologic therapy in refractory chronic non-bacterial osteomyelitis of childhood. Rheumatology (Oxford) 2010;49:1505-12. https://doi.org/10.1093/rheumatology/keq122
  49. 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. https://doi.org/10.3899/jrheum.161255
  50. Herlin T, Fiirgaard B, Bjerre M, Kerndrup G, Hasle H, Bing X, et al. Efficacy of anti-IL-1 treatment in Majeed syndrome. Ann Rheum Dis 2013;72:410-3. https://doi.org/10.1136/annrheumdis-2012-201818
  51. Hofmann SR, Kubasch AS, Range U, Laass MW, Morbach H, Girschick HJ, et al. Serum biomarkers for the diagnosis and monitoring of chronic recurrent multifocal osteomyelitis (CRMO). Rheumatol Int 2016;36:769-79. https://doi.org/10.1007/s00296-016-3466-7
  52. Miettunen PMH. Chronic non-bacterial osteitis/chronic recurrent multifocal osteomyelitis. In: Baptista MS, Tardivo JP, eds. Osteomyelitis. London: InTech; 2012:89-118.

Cited by

  1. Response of Different Treatment Protocols to Treat Chronic Non-Bacterial Osteomyelitis (CNO) of the Mandible in Adult Patients: A Systematic Review vol.17, pp.5, 2019, https://doi.org/10.3390/ijerph17051737
  2. A rare case of chronic nonbacterial osteomyelitis of the mandible in a young adult male vol.44, pp.1, 2019, https://doi.org/10.21851/obr.44.01.202003.51
  3. Pediatric Non-Infectious Osteomyelitis of the Mandible: A Case Report vol.45, pp.2, 2019, https://doi.org/10.14476/jomp.2020.45.2.39
  4. An accurate diagnosis of odontogenic cutaneous sinus tract by different computed tomography unit setting vol.47, pp.1, 2021, https://doi.org/10.5125/jkaoms.2021.47.1.51
  5. Decompression effects on bone healing in rat mandible osteomyelitis vol.11, pp.1, 2019, https://doi.org/10.1038/s41598-021-91104-7