Skip to main content
Log in

Chronic Recurrent Multifocal Osteomyelitis Associated with Chronic Inflammatory Bowel Disease in Children

  • Published:
Digestive Diseases and Sciences Aims and scope Submit manuscript

Abstract

Chronic recurrent multifocal osteomyelitis(CRMO) is a rare disease of children characterized byaseptic inflammation of the long bones and clavicles. Noinfectious etiology has been identified, and CRMO has been associated with a number of autoimmunediseases (including Wegener's granulomatosis andpsoriasis). The relationship between CRMO andinflammatory bowel disease is poorly described. Throughan internet bulletin board subscribed to by 500pediatric gastroenterologists, we identified sixinflammatory bowel disease patients (two with ulcerativecolitis, four with Crohn's colitis) with confirmed CRMO. In all cases, onset of the bony lesionspreceded the onset of bowel symptoms by as much as fiveyears. Immunosuppressive therapy for the bowel diseasegenerally resulted in improvement of the bone inflammation. Chronic recurrrent multifocalosteomyelitis should be considered in any inflammatorybowel disease patient with unexplained bone pain orareas of uptake on bone scan. CRMO may be a rareextraintestinal manifestation of inflammatory bowel disease;alternatively, certain individuals may be geneticallypredisposed to the development of bothdiseases.

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

Access this article

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

Instant access to the full article PDF.

Institutional subscriptions

Similar content being viewed by others

REFERENCES

  1. Katz J, Lichtenstein G: Rheumatologic manife stations of gastrointestinal diseases. Gastroenterol Clin North Am 27:533–562, 1998

    Google Scholar 

  2. Gokhale R, Favus M, Karrison T, Sutton M, Rich B, Kirschner B: Bone mineral density assessment in children with inflammatory bowel disease. Gastroenterology 114:902–911, 1998

    Google Scholar 

  3. Giedion A, Holthusen W, Masel L, Vischer D: Subacute and chronic" symmetrical osteomyelitis." Ann Radiol 15:329–342, 1972

    Google Scholar 

  4. Pelkonen P, Ryoppy S, Jaaskelainen J, Rapola J, Repo H, Kaitila I: Chronic osteomyelitislike disease with negative bacterial cultures. Am J Dis Child 142:1167–1173, 1988

    Google Scholar 

  5. Schuster T, Bielek J, Dietz H, Belohradsky B: Chronic recurrent multifocal osteomyelitis (CRMO). Eur J Pediatr Surg 6:45–51, 1996

    Google Scholar 

  6. Bognar M, Blake W, Agudelo C: Chronic recurrent osteomyelitis associated with Crohn's disease. Am J Med Sci 315:133–135, 1998

    Google Scholar 

  7. Sundaram M, McDonald D, Engel E, Rotman M, Siegfried E: Chronic recurrent multifocal osteomyelitis: An evolving clinical and radiological spectrum. Skeletal Radiol 25:333–336, 1996

    Google Scholar 

  8. Kotilainen P, Laxe n F, Manner I, Gullichsen R, Saario R: An aseptic inflammation of the clavicle in a patient with Crohn's disease. Scand J Rheumatol 25:112–114, 1996

    Google Scholar 

  9. Stanton R, Lopez-Sosa F, Doidge R: Chronic Recurrent Multifocal Osteomyelits. Orthop Rev 22:229–233, 1993

    Google Scholar 

  10. Hatem S, Petersilge C, Park J: Muskuloskeletal case of the day: Chronic re current multifocal osteomyelitis. AJR 169:283–292, 1997

    Google Scholar 

  11. Carr A, Cole W, Roberton D, Chow C: Chronic multifocal osteomye litis. J Bone Joint Surg 75:582–591, 1993

    Google Scholar 

  12. Byrd L, Grossmann M, Potter M, Shen-Ong G: Chronic recurrent multifocal osteomyelitis: A new recessive mutation on chromosome 18 of the mouse. Genomics 11:794–798, 1991

    Google Scholar 

  13. Prose N, Fahrner L, Miller C, Layfield L: Pustular psoriasis with chronic recurrent multifocal osteomye litis and spontaneous fractures. J Am Acad Dermatol 31:376–379, 1994

    Google Scholar 

  14. Paller A, Pachman L, Rich K, Esterly N, Gonzalez-Crussi F: Pustulosis palmaris et plantaris: Its association with chronic recurrent multifocal osteomyelitis. J Am Acad De rmatol 12:927–930, 1985

    Google Scholar 

  15. Majeed H, Kalaawi M, Mohanty D, Teebi A, Tunjekar M, Al-Gharbawy F, Majeed S, Al-Gazzar A: Chronic dyserythropoietic anemia and recurrent multifocal osteomyelitis in three related children and the association with Sweet syndrome in two siblings. J Pediatr 115:730–734, 1989

    Google Scholar 

  16. Dagan O, Barak Y, Metzker A: Pyoderma gangrenosum and sterile multifocal osteomyelitis preceding the appearance of Takayasu arteritis. Pediatr Dermatol 12:39–42, 1995

    Google Scholar 

  17. Kerem E, Manson D, Laxer R, Levison H, Reilly B: Pulmonary association in a case of chronic recurrent multifocal osteomyelitis. Pediatr Pulmonol 7:55–58, 1989

    Google Scholar 

  18. Kahn M, Bouchon J, Charnot A, Palazzo E: Enteropathies chroniques et syndrome SAPHO. 8 Observations. Rev Rheum Mal Osteoartic 59:91–94, 1992

    Google Scholar 

  19. Andersson R: Effective treatment with interferon-alpha in chronic recurrent multifocal osteomyelitis. J Interferon Cytokine Res 15:837–838, 1995

    Google Scholar 

  20. Gallagher K, Roberts R, MacFarlane A, Stiehm E: Treatment of chronic recurrent multifocal osteomyelitis with interferon gamma. J Pediatr 131:470–472, 1997

    Google Scholar 

  21. Sartor R: Current concepts in the etiology and pathogenesis of ulcerative colitis and Crohn' s disease. Gastroenterol Clin North Am 24:475–507, 1995

    Google Scholar 

  22. Camoglio L, TeVelde A, Tigges A, Das P, VanDeventer S: Altered expression of interferon-gamma and interleukin-4 in inflammatory bowel disease. Inflamm Bowel Dis 4:285–290, 1998

    Google Scholar 

  23. Bross D, Leichtner A, Zurakowski D, Law T, Bousvaros A: Elevation of serum interleukin-6 but not serum interleukin 2 receptor in children with Crohn's disease. J Pediatr Gastroenterol Nutr 23:164–171, 1996

    Google Scholar 

  24. Proujansky R, Carpenter A: Soluble interleukin-2 receptor as a marker of lymphocyte activation in childhood Crohn's disease. J Pediatr Gastroenterol Nutr 13:277–284, 1991

    Google Scholar 

  25. Hyams J, Fitzgerald J, Treem W, Wyzga N, Kreutzer D: Relationship of functional and antigenic interleukin-6 to disease activity in inflammatory bowel disease. Gastroenterology 104:1285–1292, 1993

    Google Scholar 

  26. Steheli L, Church C, Ward B: Infantile cortical hyperostosis (Caffey' s disease). JAMA 203:384, 1968

    Google Scholar 

  27. Heyman E, Laver J, Beer S: Prostaglandin synthetase inhibitor in Caffey disease. J Pediatr 101:314, 1982 ( letter)

    Google Scholar 

  28. Poznanski A, Feinbach S, Berry T: Bone changes from prostaglandin therapy. Skeletal Radiology 14:20–25, 1985

    Google Scholar 

  29. Kozawa O, Tokuda H, Kaida T, Matsuno H, Uematsu T: Effect of vitamin D3 on interleukin-6 synthesis induced by prostaglandins in osteoblasts. Leukotrienes Essent Fatty Acids 58:119–123, 1998

    Google Scholar 

  30. Freeman H: Osteomyelitis and osteonecrosis in inflammatory bowel disease. Can J Gastroenterol 11:601–606, 1997

    Google Scholar 

  31. Neale G, Kelsall A, Doyle F: Crohn's disease and diffuse symmetrical periostitis. Gut 9:383–387, 1968

    Google Scholar 

  32. Martin J, Desvosa R, O'Sullivan M, Silverstone E, Williams H: Chronic recurrent multifocal osteomye litis: Spinal involvement and radiological appearances. Br J Rheumatol 35:1019–1021, 1996

    Google Scholar 

Download references

Authors

Rights and permissions

Reprints and permissions

About this article

Cite this article

Bousvaros, A., Marcon, M., Treem, W. et al. Chronic Recurrent Multifocal Osteomyelitis Associated with Chronic Inflammatory Bowel Disease in Children. Dig Dis Sci 44, 2500–2507 (1999). https://doi.org/10.1023/A:1026695224019

Download citation

  • Issue Date:

  • DOI: https://doi.org/10.1023/A:1026695224019

Navigation