Skip to main content

Advertisement

SpringerLink
Log in

Complete resolution and remodeling of chronic recurrent multifocal osteomyelitis on MRI and radiographs

  • Case Report
  • Published:
Skeletal Radiology Aims and scope Submit manuscript

Abstract

Chronic recurrent multifocal osteomyelitis (CRMO) is a rare condition thought to be under-diagnosed, with a true prevalence of more than the 1 in 10,000 estimated. It is a condition that is classically described as polyostotic with a relapsing and remitting course, preferentially affecting the metaphyses of tubular bones in the pediatric population. Lesions have characteristic appearances of cortical hyperostosis and mixed lytic/sclerotic medullary appearances radiographically, with active osteitis and periostitis best seen with fluid-sensitive sequences on magnetic resonance imaging (MRI). There are reports of lesions resolving on follow-up radiographs and MRI scans, but no supporting images. In particular, although the marrow appearances and degree of osteitis have been shown to improve on MRI, complete resolution and remodeling back to normal has never been demonstrated. We present a case of a lesion that has completely healed and remodeled back to normal appearances on both radiographs and MRI, and consider this the standard for the often loosely used terms “normalization” and “resolution”. We discuss the implications of this for our understanding of the natural history of CRMO, and how this adds weight to the condition being significantly under-diagnosed. It provides a “gold standard” to be aimed for when assessing treatments for CRMO, and the optimal outcomes that are possible. It also provides further insight into the potential of pediatric bone to recover and remodel when affected by inflammatory conditions.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3

Similar content being viewed by others

References

  1. Fritz J, Tzaribatchev N, Claussen CD, Carrino JA, Horger MS. Chronic recurrent multifocal osteomyelitis: comparison of whole-body MR imaging with radiography and correlation with clinical and laboratory data. Radiology. 2009;252(3):842–51.

    PubMed  Google Scholar 

  2. Jurik AG, Egund N. MRI in chronic recurrent multifocal osteomyelitis. Skeletal Radiol. 1997;26(4):230–8.

    CAS  PubMed  Google Scholar 

  3. Giedion A, Holthusen W, Masel LF, Vischer D. Subacute and chronic “symmetrical” osteomyelitis. Ann Radiol (Paris). 1972;15(3):329–42.

    CAS  Google Scholar 

  4. Probst FP, Björksten B, Gustavson KH. Radiological aspect of chronic recurrent multifocal osteomyelitis. Ann Radiol (Paris). 1978;21(2–3):115–25.

    CAS  Google Scholar 

  5. Gustavson KH, Wilbrand HF. Chronic symmetric osteomyelitis. Report of a case. Acta Radiol Diagn (Stockh). 1974;15(5):551–7.

    CAS  Google Scholar 

  6. Jansson A, Renner ED, Ramser J, Mayer A, Haban M, Meindl A, et al. Classification of non-bacterial osteitis: retrospective study of clinical, immunological and genetic aspects in 89 patients. Rheumatol Oxf Engl. 2007;46(1):154–60.

    CAS  Google Scholar 

  7. Jurik AG, Helmig O, Ternowitz T, Møller BN. Chronic recurrent multifocal osteomyelitis: a follow-up study. J Pediatr Orthop. 1988;8(1):49–58.

    CAS  PubMed  Google Scholar 

  8. Wipff J, Costantino F, Lemelle I, Pajot C, Duquesne A, Lorrot M, et al. A large national cohort of French patients with chronic recurrent multifocal osteitis: prognostic factors, outcomes, and management of CRMO. Arthritis Rheumatol. 2015;67(4):1128–37.

    CAS  PubMed  Google Scholar 

  9. Duffy CM, Lam PY, Ditchfield M, Allen R, Graham HK. Chronic recurrent multifocal osteomyelitis: review of orthopaedic complications at maturity. J Pediatr Orthop. 2002;22(4):501–5.

    PubMed  Google Scholar 

  10. Carr AJ, Cole WG, Roberton DM, Chow CW. Chronic multifocal osteomyelitis. J Bone Joint Surg Br. 1993;75(4):582–91.

    CAS  PubMed  Google Scholar 

  11. 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.

    CAS  PubMed  Google Scholar 

  12. Roderick M, Shah R, Finn A, Ramanan AV. Efficacy of pamidronate therapy in children with chronic non-bacterial osteitis: disease activity assessment by whole body magnetic resonance imaging. Rheumatology. 2014;53(11):1973–6.

    CAS  PubMed  Google Scholar 

  13. Iyer RS, Thapa MM, Chew FS. Chronic recurrent multifocal osteomyelitis: review. AJR Am J Roentgenol. 2011;196(6 Suppl):S87–91.

    PubMed  Google Scholar 

  14. Witt M, Meier J, Hammitzsch A, Proft F, Schulze-Koops H, Grunke M. Disease burden, disease manifestations and current treatment regimen of the SAPHO syndrome in Germany: results from a nationwide patient survey. Semin Arthritis Rheum. 2014;43(6):745–50.

    PubMed  Google Scholar 

  15. Karadag-Saygi E, Gunduz OH, Gumrukcu G, Akyuz G. SAPHO syndrome: misdiagnosed and operated. Acta Reumatol Port. 2008;33(4):460–3.

    PubMed  Google Scholar 

  16. Falip C, Alison M, Boutry N, Job-Deslandre C, Cotten A, Azoulay R, et al. Chronic recurrent multifocal osteomyelitis (CRMO): a longitudinal case series review. Pediatr Radiol. 2013;43(3):355–75.

    PubMed  Google Scholar 

  17. Depasquale R, Kumar N, Lalam RK, Tins BJ, Tyrrell PNM, Singh J, et al. SAPHO: what radiologists should know. Clin Radiol. 2012;67(3):195–206.

    CAS  PubMed  Google Scholar 

  18. Leone A, Cassar-Pullicino VN, Casale R, Magarelli N, Semprini A, Colosimo C. The SAPHO syndrome revisited with an emphasis on spinal manifestations. Skeletal Radiol. 2015;44(1):9–24.

    PubMed  Google Scholar 

  19. Kaiser D, Bolt I, Hofer M, Relly C, Berthet G, Bolz D, et al. Chronic nonbacterial osteomyelitis in children: a retrospective multicenter study. Pediatr Rheumatol Online J. 2015;13:25. https://doi.org/10.1186/s12969-015-0023-y

    Article  PubMed  PubMed Central  Google Scholar 

  20. Court C, Charlez C, Molina V, Clerc D, Miquel A, Nordin JY. Isolated thoracic spine lesion: is this the presentation of a SAPHO syndrome? A case report. Eur Spine J. 2005;14(7):711–5.

    PubMed  Google Scholar 

  21. Bjorksten B, Boquist L. Histopathological aspects of chronic recurrent multifocal osteomyelitis. J Bone Joint Surg Br. 1980;62(3):376–80.

    Google Scholar 

  22. Beck-Broichsitter BE, Smeets R, Heiland M. Current concepts in pathogenesis of acute and chronic osteomyelitis. Curr Opin Infect Dis. 2015;28(3):240–5.

    CAS  PubMed  Google Scholar 

  23. Yu L, Kasser JR, O’Rourke E, Kozakewich H. Chronic recurrent multifocal osteomyelitis. Association with vertebra plana. J Bone Joint Surg Am. 1989;71(1):105–12.

    CAS  PubMed  Google Scholar 

  24. Mandell GA, Contreras SJ, Conard K, Harcke HT, Maas KW. Bone scintigraphy in the detection of chronic recurrent multifocal osteomyelitis. J Nucl Med. 1998;39(10):1778–83.

    CAS  PubMed  Google Scholar 

  25. Acikgoz G, Averill LW. Chronic recurrent multifocal osteomyelitis: typical patterns of bone involvement in whole-body bone scintigraphy. Nucl Med Commun 2014;35(8):797–807.

    PubMed  Google Scholar 

  26. Surendra G, Shetty U. Chronic recurrent multifocal osteomyelitis: a rare entity: CRMO: a rare entity. J Med Imaging Radiat Oncol. 2015;59(4):436–44.

    PubMed  Google Scholar 

  27. Fritz J. The contributions of whole-body magnetic resonance imaging for the diagnosis and management of chronic recurrent multifocal osteomyelitis. J Rheumatol. 2015;42(8):1359–60.

    CAS  PubMed  Google Scholar 

  28. Guérin-Pfyffer S, Guillaume-Czitrom S, Tammam S, Koné-Paut I. Evaluation of chronic recurrent multifocal osteitis in children by whole-body magnetic resonance imaging. Jt Bone Spine Rev Rhum. 2012;79(6):616–20.

    Google Scholar 

  29. 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.

    PubMed  Google Scholar 

  30. Zhao Y, Dedeoglu F, Ferguson PJ, Lapidus SK, Laxer RM, Bradford MC, et al. Physicians’ perspectives on the diagnosis and treatment of chronic nonbacterial osteomyelitis. Int J Rheumatol. 2017;2017:7694942.

    PubMed  PubMed Central  Google Scholar 

  31. Dangman BC, Hoffer FA, Rand FF, O’Rourke EJ. Osteomyelitis in children: gadolinium-enhanced MR imaging. Radiology. 1992;182(3):743–7.

    CAS  PubMed  Google Scholar 

  32. Hayem G, Bouchaud-Chabot A, Benali K, Roux S, Palazzo E, Silbermann-Hoffman O, et al. SAPHO syndrome: a long-term follow-up study of 120 cases. Semin Arthritis Rheum. 1999;29(3):159–71.

    CAS  PubMed  Google Scholar 

  33. Hermann K-GA, Bollow M. Magnetic resonance imaging of sacroiliitis in patients with spondyloarthritis: correlation with anatomy and histology. Rofo. 2014;186(3):230–7.

    PubMed  Google Scholar 

  34. Van Onna M, van Tubergen A, van der Heijde DM, Jurik AG, Landewé R. Bone marrow edema on magnetic resonance imaging (MRI) of the sacroiliac joints is associated with development of fatty lesions on MRI over a 1-year interval in patients with early inflammatory low back pain: a 2-year followup study. J Rheumatol 2014;41(6):1088–1094.

    PubMed  Google Scholar 

  35. Wilkins KE. Principles of fracture remodeling in children. Injury. 2005;36(Suppl 1):A3–11.

    PubMed  Google Scholar 

  36. Lindaman LM. Bone healing in children. Clin Podiatr Med Surg. 2001;18(1):97–108.

    CAS  PubMed  Google Scholar 

  37. Caffey J. Infantile cortical hyperostoses. J Pediatr. 1946;29(5):541–59.

    CAS  PubMed  Google Scholar 

  38. Kutty N, Thomas D, George L, John TB. Caffey disease or infantile cortical hyperostosis: a case report. Oman Med J. 2010;25(2):134–6.

    PubMed  PubMed Central  Google Scholar 

  39. Navarre P, Pehlivanov I, Morin B. Recurrence of infantile cortical hyperostosis: a case report and review of the literature. J Pediatr Orthop. 2013;33(2):e10–7.

    PubMed  Google Scholar 

  40. Caffey J, Silverman WA. Infantile cortical hyperostosis. Preliminary report on a new syndrome. Am J Roentgenol. 1945;54:1–16.

    Google Scholar 

  41. Sanders DG, Weijers RE. MRI findings in Caffey’s disease. Pediatr Radiol. 1994;24(5):325–7.

    CAS  PubMed  Google Scholar 

  42. Colina M, Govoni M, Orzincolo C, Trotta F. Clinical and radiologic evolution of synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: a single center study of a cohort of 71 subjects. Arthritis Rheum. 2009;61(6):813–21.

    PubMed  Google Scholar 

  43. Matzaroglou C, Velissaris D, Karageorgos A, Marangos M, Panagiotopoulos E, Karanikolas M. SAPHO syndrome diagnosis and treatment: report of five cases and review of the literature. Open Orthop J. 2009;3:100–6.

    PubMed  PubMed Central  Google Scholar 

  44. Altman AR, Tseng W-J, de Bakker CMJ, Chandra A, Lan S, Huh BK, et al. Quantification of skeletal growth, modeling, and remodeling by in vivo micro computed tomography. Bone 2015;81:370–379.

    PubMed  PubMed Central  Google Scholar 

  45. Prentice A, Schoenmakers I, Laskey MA, de Bono S, Ginty F, Goldberg GR. Nutrition and bone growth and development. Proc Nutr Soc 2006;65(4):348–360.

    CAS  PubMed  PubMed Central  Google Scholar 

Download references

Acknowledgements

We would like to thank our colleagues at the Robert Jones and Agnes Hunt Orthopedic Hospital.

Funding

No funding received.

Author information

Authors and Affiliations

  1. Department of Diagnostic Imaging, Robert Jones and Agnes Hunt Orthopaedic Hospital NHS Foundation Trust, Oswestry, Shropshire, SY10 7AG, UK

    Y. J. Berkowitz, S. J. Greenwood & V. N. Cassar-Pullicino

  2. Department of Orthopaedic Oncology, Robert Jones and Agnes Hunt Orthopaedic Hospital NHS Foundation Trust, Oswestry, Shropshire, SY10 7AG, UK

    G. Cribb

  3. Department of Medicine, Robert Jones and Agnes Hunt Orthopaedic Hospital NHS Foundation Trust, Oswestry, Shropshire, SY10 7AG, UK

    K. Davies

Authors
  1. Y. J. Berkowitz
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. S. J. Greenwood
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. G. Cribb
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. K. Davies
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. V. N. Cassar-Pullicino
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to V. N. Cassar-Pullicino.

Ethics declarations

Conflicts of interest

The authors declare that they have no conflicts of interest.

Ethical approval

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Declaration of Helsinki and its later amendments or comparable ethical standards.

Informed consent

Informed consent was obtained from the patient and her legal guardians.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Berkowitz, Y.J., Greenwood, S.J., Cribb, G. et al. Complete resolution and remodeling of chronic recurrent multifocal osteomyelitis on MRI and radiographs. Skeletal Radiol 47, 563–568 (2018). https://doi.org/10.1007/s00256-017-2812-5

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00256-017-2812-5

Keywords

Search

Navigation