Adult-onset Chronic Recurrent Multifocal Osteomyelitis with High Intensity of Muscles Detected by Magnetic Resonance Imaging, Successfully Controlled with Tocilizumab
Chronic recurrent multifocal osteomyelitis (CRMO) is an autoinflammatory bone disorder that generally occurs in children and predominantly affects the long bones with marginal sclerosis. We herein report two cases of adult-onset CRMO involving the tibial diaphysis bilaterally, accompanied by polyart...
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| Published in | Internal Medicine Vol. 56; no. 17; pp. 2353 - 2360 |
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| Main Authors | , , , , , , , , , , , |
| Format | Journal Article |
| Language | English |
| Published |
Japan
The Japanese Society of Internal Medicine
01.01.2017
Japan Science and Technology Agency |
| Subjects | |
| Online Access | Get full text |
| ISSN | 0918-2918 1349-7235 1349-7235 |
| DOI | 10.2169/internalmedicine.8473-16 |
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| Abstract | Chronic recurrent multifocal osteomyelitis (CRMO) is an autoinflammatory bone disorder that generally occurs in children and predominantly affects the long bones with marginal sclerosis. We herein report two cases of adult-onset CRMO involving the tibial diaphysis bilaterally, accompanied by polyarthritis. Magnetic resonance imaging (MRI) showed both tibial osteomyelitis and high intensity of the extensive lower leg muscles. Anti-interleukin-6 therapy with tocilizumab (TCZ) effectively controlled symptoms and inflammatory markers in both patients. High intensity of the lower leg muscles detected by MRI also improved. These cases demonstrate that CRMO should be included in the differential diagnosis of adult patients with bone pain, inflammation, and high intensity of the muscles detected by MRI. TCZ may therefore be an effective therapy for muscle inflammation of CRMO. |
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| AbstractList | Chronic recurrent multifocal osteomyelitis (CRMO) is an autoinflammatory bone disorder that generally occurs in children and predominantly affects the long bones with marginal sclerosis. We herein report two cases of adult-onset CRMO involving the tibial diaphysis bilaterally, accompanied by polyarthritis. Magnetic resonance imaging (MRI) showed both tibial osteomyelitis and high intensity of the extensive lower leg muscles. Anti-interleukin-6 therapy with tocilizumab (TCZ) effectively controlled symptoms and inflammatory markers in both patients. High intensity of the lower leg muscles detected by MRI also improved. These cases demonstrate that CRMO should be included in the differential diagnosis of adult patients with bone pain, inflammation, and high intensity of the muscles detected by MRI. TCZ may therefore be an effective therapy for muscle inflammation of CRMO. Chronic recurrent multifocal osteomyelitis (CRMO) is an autoinflammatory bone disorder that generally occurs in children and predominantly affects the long bones with marginal sclerosis. We herein report two cases of adult-onset CRMO involving the tibial diaphysis bilaterally, accompanied by polyarthritis. Magnetic resonance imaging (MRI) showed both tibial osteomyelitis and high intensity of the extensive lower leg muscles. Anti-interleukin-6 therapy with tocilizumab (TCZ) effectively controlled symptoms and inflammatory markers in both patients. High intensity of the lower leg muscles detected by MRI also improved. These cases demonstrate that CRMO should be included in the differential diagnosis of adult patients with bone pain, inflammation, and high intensity of the muscles detected by MRI. TCZ may therefore be an effective therapy for muscle inflammation of CRMO.Chronic recurrent multifocal osteomyelitis (CRMO) is an autoinflammatory bone disorder that generally occurs in children and predominantly affects the long bones with marginal sclerosis. We herein report two cases of adult-onset CRMO involving the tibial diaphysis bilaterally, accompanied by polyarthritis. Magnetic resonance imaging (MRI) showed both tibial osteomyelitis and high intensity of the extensive lower leg muscles. Anti-interleukin-6 therapy with tocilizumab (TCZ) effectively controlled symptoms and inflammatory markers in both patients. High intensity of the lower leg muscles detected by MRI also improved. These cases demonstrate that CRMO should be included in the differential diagnosis of adult patients with bone pain, inflammation, and high intensity of the muscles detected by MRI. TCZ may therefore be an effective therapy for muscle inflammation of CRMO. |
| Author | Ito, Tomoyuki Wada, Yoko Narita, Ichiei Hasegawa, Eriko Saeki, Takako Nozawa, Yukiko Suzuki, Yoshiki Sato, Hiroe Kuroda, Takeshi Umezu, Hajime Nakano, Masaaki Nakatsue, Takeshi |
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| BackLink | https://www.ncbi.nlm.nih.gov/pubmed/28794369$$D View this record in MEDLINE/PubMed |
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| Keywords | osteomyelitis tocilizumab polyarthritis chronic recurrent multifocal osteitis (CRMO) SAPHO syndrome myositis |
| Language | English |
| License | The Internal Medicine is an Open Access article distributed under the Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License. To view the details of this license, please visit (https://creativecommons.org/licenses/by-nc-nd/4.0/). |
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| References | 16. Nagashima S, Nozawa T, Kizawa T, et al. [Chronic recurrent multifocal osteomyelitis with interstitial myositis]. Nihon Rinsho Meneki Gakkai Kaishi (Jpn J Clin Immunol) 36: 52-57, 2013 (in Japanese, Abstract in English). 6. Nguyen MT, Borchers A, Selmi C, Naguwa SM, Cheema G, Gershwin ME. The SAPHO syndrome. Semin Arthritis Rheum 42: 254-265, 2012. 26. Hamanoue S, Suwabe T, Hoshino J, et al. Successful treatment with humanized anti-interleukin-6 receptor antibody (tocilizumab) in a case of AA amyloidosis complicated by familial Mediterranean fever. Mod Rheumatol 26: 610-613, 2016. 18. Calguneri M, Apras S, Ozbalkan Z, Ozturk MA. The efficacy of interferon-alpha in a patient with resistant familial Mediterranean fever complicated by polyarteritis nodosa. Intern Med 43: 612-614, 2004. 13. Beck C, Morbach H, Beer M, et al. Chronic nonbacterial osteomyelitis in childhood: prospective follow-up during the first year of anti-inflammatory treatment. Arthritis Res Ther 12: R74, 2010. 22. Hofmann SR, Roesen-Wolff A, Hahn G, Hedrich CM. Update: cytokine dysregulation in chronic nonbacterial osteomyelitis (CNO). Int J Rheumatol 2012: 310206, 2012. 27. Hosoya T, Mizoguchi F, Hasegawa H, et al. A case presenting with the clinical characteristics of tumor necrosis factor (TNF) receptor-associated periodic syndrome (TRAPS) without TNFRSF1A mutations successfully treated with tocilizumab. Intern Med 54: 2069-2072, 2015. 14. Benhamou CL, Chamot AM, Kahn MF. Synovitis-acne-pustulosis hyperostosis-osteomyelitis syndrome (SAPHO). A new syndrome among the spondyloarthropathies? Clin Exp Rheumatol 6: 109-112, 1988. 2. Stern SM, Ferguson PJ. Autoinflammatory bone diseases. Rheum Dis Clin North Am 39: 735-749, 2013. 3. Borzutzky A, Stern S, Reiff A, et al. Pediatric chronic nonbacterial osteomyelitis. Pediatrics 130: e1190-e1197, 2012. 20. 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 11: 47, 2013. 21. Hofmann SR, Schwarz T, Moller JC, et al. Chronic non-bacterial osteomyelitis is associated with impaired Sp1 signaling, reduced IL10 promoter phosphorylation, and reduced myeloid IL-10 expression. Clin Immunol 141: 317-327, 2011. 4. Khanna G, Sato TS, Ferguson P. Imaging of chronic recurrent multifocal osteomyelitis. Radiographics 29: 1159-1177, 2009. 17. Komatsu S, Honma M, Igawa S, et al. Cutaneous necrotizing vasculitis as a manifestation of familial Mediterranean fever. J Dermatol 41: 827-829, 2014. 10. Surendra G, Shetty U. Chronic recurrent multifocal osteomyelitis: a rare entity. J Med Imaging Radiat Oncol 59: 436-444, 2015. 8. Hong CW, Hsiao EC, Horvai AE, Link TM. Chronic recurrent multifocal osteomyelitis with an atypical presentation in an adult man. Skeletal Radiol 44: 1359-1364, 2015. 19. Golla A, Jansson A, Ramser J, et al. Chronic recurrent multifocal osteomyelitis (CRMO): evidence for a susceptibility gene located on chromosome 18q21.3-18q22. Eur J Hum Genet 10: 217-221, 2002. 23. Simm PJ, Allen RC, Zacharin MR. Bisphosphonate treatment in chronic recurrent multifocal osteomyelitis. J Pediatr 152: 571-575, 2008. 1. Wipff J, Costantino F, Lemelle I, et al. A large national cohort of French patients with chronic recurrent multifocal osteitis. Arthritis Rheumatol 67: 1128-1137, 2015. 7. Chow LT, Griffith JF, Kumta SM, Leung PC. Chronic recurrent multifocal osteomyelitis: a great clinical and radiologic mimic in need of recognition by the pathologist. APMIS 107: 369-379, 1999. 25. Koga T, Migita K, Kawakami A. Biologic therapy in familial Mediterranean fever. Mod Rheumatol 26: 637-641, 2016. 15. Guerin-Pfyffer S, Guillaume-Czitrom S, Tammam S, Kone-Paut I. Evaluation of chronic recurrent multifocal osteitis in children by whole-body magnetic resonance imaging. Joint Bone Spine 79: 616-620, 2012. 5. Jansson AF, Muller TH, Gliera L, et al. Clinical score for nonbacterial osteitis in children and adults. Arthritis Rheum 60: 1152-1159, 2009. 11. Wipff J, Adamsbaum C, Kahan A, Job-Deslandre C. Chronic recurrent multifocal osteomyelitis. Joint Bone Spine 78: 555-560, 2011. 9. Takeuchi K, Matsusita M, Takagishi K. A case of SAPHO (synovitis-acne-pustulosis-hyperostosis-osteomyelitis) syndrome in which [18F]fluorodeoxyglucose positron emission tomography was useful for differentiating from multiple metastatic bone tumors. Mod Rheumatol 17: 67-71, 2007. 12. 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 3: 733-738, 2007. 24. 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. 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| References_xml | – reference: 12. 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 3: 733-738, 2007. – reference: 20. 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 11: 47, 2013. – reference: 15. Guerin-Pfyffer S, Guillaume-Czitrom S, Tammam S, Kone-Paut I. Evaluation of chronic recurrent multifocal osteitis in children by whole-body magnetic resonance imaging. Joint Bone Spine 79: 616-620, 2012. – reference: 22. Hofmann SR, Roesen-Wolff A, Hahn G, Hedrich CM. Update: cytokine dysregulation in chronic nonbacterial osteomyelitis (CNO). Int J Rheumatol 2012: 310206, 2012. – reference: 17. Komatsu S, Honma M, Igawa S, et al. Cutaneous necrotizing vasculitis as a manifestation of familial Mediterranean fever. J Dermatol 41: 827-829, 2014. – reference: 16. Nagashima S, Nozawa T, Kizawa T, et al. [Chronic recurrent multifocal osteomyelitis with interstitial myositis]. Nihon Rinsho Meneki Gakkai Kaishi (Jpn J Clin Immunol) 36: 52-57, 2013 (in Japanese, Abstract in English). – reference: 9. Takeuchi K, Matsusita M, Takagishi K. A case of SAPHO (synovitis-acne-pustulosis-hyperostosis-osteomyelitis) syndrome in which [18F]fluorodeoxyglucose positron emission tomography was useful for differentiating from multiple metastatic bone tumors. Mod Rheumatol 17: 67-71, 2007. – reference: 8. Hong CW, Hsiao EC, Horvai AE, Link TM. Chronic recurrent multifocal osteomyelitis with an atypical presentation in an adult man. Skeletal Radiol 44: 1359-1364, 2015. – reference: 14. Benhamou CL, Chamot AM, Kahn MF. Synovitis-acne-pustulosis hyperostosis-osteomyelitis syndrome (SAPHO). A new syndrome among the spondyloarthropathies? Clin Exp Rheumatol 6: 109-112, 1988. – reference: 18. Calguneri M, Apras S, Ozbalkan Z, Ozturk MA. The efficacy of interferon-alpha in a patient with resistant familial Mediterranean fever complicated by polyarteritis nodosa. Intern Med 43: 612-614, 2004. – reference: 23. Simm PJ, Allen RC, Zacharin MR. Bisphosphonate treatment in chronic recurrent multifocal osteomyelitis. J Pediatr 152: 571-575, 2008. – reference: 10. Surendra G, Shetty U. Chronic recurrent multifocal osteomyelitis: a rare entity. J Med Imaging Radiat Oncol 59: 436-444, 2015. – reference: 3. Borzutzky A, Stern S, Reiff A, et al. Pediatric chronic nonbacterial osteomyelitis. Pediatrics 130: e1190-e1197, 2012. – reference: 7. Chow LT, Griffith JF, Kumta SM, Leung PC. Chronic recurrent multifocal osteomyelitis: a great clinical and radiologic mimic in need of recognition by the pathologist. APMIS 107: 369-379, 1999. – reference: 2. Stern SM, Ferguson PJ. Autoinflammatory bone diseases. Rheum Dis Clin North Am 39: 735-749, 2013. – reference: 25. Koga T, Migita K, Kawakami A. Biologic therapy in familial Mediterranean fever. Mod Rheumatol 26: 637-641, 2016. – reference: 27. Hosoya T, Mizoguchi F, Hasegawa H, et al. A case presenting with the clinical characteristics of tumor necrosis factor (TNF) receptor-associated periodic syndrome (TRAPS) without TNFRSF1A mutations successfully treated with tocilizumab. Intern Med 54: 2069-2072, 2015. – reference: 1. Wipff J, Costantino F, Lemelle I, et al. A large national cohort of French patients with chronic recurrent multifocal osteitis. Arthritis Rheumatol 67: 1128-1137, 2015. – reference: 6. Nguyen MT, Borchers A, Selmi C, Naguwa SM, Cheema G, Gershwin ME. The SAPHO syndrome. Semin Arthritis Rheum 42: 254-265, 2012. – reference: 26. Hamanoue S, Suwabe T, Hoshino J, et al. Successful treatment with humanized anti-interleukin-6 receptor antibody (tocilizumab) in a case of AA amyloidosis complicated by familial Mediterranean fever. Mod Rheumatol 26: 610-613, 2016. – reference: 4. 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| Title | Adult-onset Chronic Recurrent Multifocal Osteomyelitis with High Intensity of Muscles Detected by Magnetic Resonance Imaging, Successfully Controlled with Tocilizumab |
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