Abstract
There are only a few studies that address the frequency and type of spinal involvement in patients with chronic recurrent multifocal osteomyelitis (CRMO) as well as the outcome of these patients treated with pamidronate (PAM). We performed a retrospective study on patients with CRMO and analyzed clinical and pain assessments as well as regional and whole body MRI findings and compared with posttreatment findings. Of 102 children and adolescents with CRMO, 27 (26%) had involvement of the spine. Vertebral deformities were seen in 14 of these 27 patients, scoliosis or kyphosis in 6. After routine whole body MRI, 19 complained of back pain, whereas eight were asymptomatic with spinal lesions detected incidentally. A total of 72 spinal lesions were detected, thoracic vertebrae being the most commonly affected. Seven patients were treated with PAM; all of whom had vertebral deformities and ongoing back pain. Pain resolution was achieved within 3 months of PAM treatment in every case. One patient subsequently developed a pain amplification syndrome. Repeat MRI performed at a mean interval of 13 months revealed partial or complete resolution of vertebral hyperintensities in every patient. Improvement of vertebral height was seen in a total of three vertebrae in two patients. Severe side effects were not observed. In conclusion, we demonstrated that spinal involvement and associated vertebral deformities with or without kyphoscoliosis are not rare in CRMO, and PAM appears to be an effective and safe treatment for this condition. Although controlled studies are urgently needed, the use of PAM for refractory CRMO with extended spinal involvement (vertebral deformities, kyphosis, and scoliosis) should be considered, especially after failing of conventional therapy.
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Abbreviations
- CRMO:
-
chronic recurrent multifocal osteomyelitis
- NSAID:
-
nonsteroidal anti-inflammatory drug
- PAM:
-
pamidronate
- TNF α:
-
tumor necrosis factor alpha
References
Anderson SE, Heini P, Sauvain MJ et al (2003) Imaging of chronic recurrent multifocal osteomyelitis of childhood first presenting with isolated primary spinal involvement. Skeletal Radiol 32(6):328–336
Bjorksten B, Boquist L (1980) Histopathological aspects of chronic recurrent multifocal osteomyelitis. J Bone Joint Surg Br 62(3):376–380
Carr AJ, Cole WG, Roberton DM et al (1993) Chronic multifocal osteomyelitis. J Bone Joint Surg Br 75(4):582–591
Catalano-Pons C, Comte A, Wipff J et al (2008) Clinical outcome in children with chronic recurrent multifocal osteomyelitis. Rheumatology (Oxford) 47(9):1397–1399
Compeyrot-Lacassagne S, Rosenberg AM, Babyn P et al (2007) Pamidronate treatment of chronic noninfectious inflammatory lesions of the mandible in children. J Rheumatol 34(7):1585–1589
Corrado A, Santoro N, Cantatore FP (2007) Extra-skeletal effects of bisphosphonates. Joint Bone Spine 74(1):32–38
De Cunto A, Maschio M, Lepore L et al (2009) A case of chronic recurrent multifocal osteomyelitis successfully treated with neridronate. J Pediatr 154(1):154–155, author reply 155
Deogaonkar K, Ghandour A, Jones A et al (2008) Chronic recurrent multifocal osteomyelitis presenting as acute scoliosis: a case report and review of literature. Eur Spine J 17(Suppl 2):S248–2S52
Deutschmann A, Mache CJ, Bodo K et al (2005) Successful treatment of chronic recurrent multifocal osteomyelitis with tumor necrosis factor-alpha blockage. Pediatrics 116(5):1231–1233
Duffy CM, Lam PY, Ditchfield M et al (2002) Chronic recurrent multifocal osteomyelitis: review of orthopaedic complications at maturity. J Pediatr Orthop 22(4):501–505
Giedion A, HolthusenW MLF et al (1972) Subacute and chronic “symmetrical” osteomyelitis. Ann Radiol (Paris) 15(3):329–342
Girschick HJ, Raab P, Surbaum S et al (2005) Chronic non-bacterial osteomyelitis in children. Ann Rheum Dis 64(2):279–285
Gleeson H, Wiltshire E, Briody J et al (2008) Childhood chronic recurrent multifocal osteomyelitis: pamidronate therapy decreases pain and improves vertebral shape. J Rheumatol 35(4):707–712
Hayem G, Bouchaud-Chabot A, Benali K et al (1999) SAPHO syndrome: a long-term follow-up study of 120 cases. Semin Arthritis Rheum 29(3):159–171
Huber AM, Lam PY, Duffy CM et al (2002) Chronic recurrent multifocal osteomyelitis: clinical outcomes after more than five years of follow-up. J Pediatr 141(2):198–203
Ishikawa-Nakayama K, Sugiyama E, Sawazaki S et al (2000) Chronic recurrent multifocal osteomyelitis showing marked improvement with corticosteroid treatment. J Rheumatol 27(5):1318–1319
Jansson A, Renner ED, Ramser J et al (2007) Classification of non-bacterial osteitis: retrospective study of clinical, immunological and genetic aspects in 89 patients. Rheumatology (Oxford) 46(1):154–160
Job-Deslandre C, Krebs S, Kahan A (2001) Chronic recurrent multifocal osteomyelitis: five-year outcomes in 14 pediatric cases. Joint Bone Spine 68(3):245–251
Jurik AG (2004) Chronic recurrent multifocal osteomyelitis. Semin Musculoskelet Radiol 8(3):243–253
Jurik AG, Egund N (1997) MRI in chronic recurrent multifocal osteomyelitis. Skeletal Radiol 26(4):230–238
Kayani I, Syed I, Saifuddin A et al (2004) Vertebral osteomyelitis without disc involvement. Clin Radiol 59(10):881–891
Kellenberger CJ, Epelman M, Miller SF et al (2004) Fast STIR whole-body MR imaging in children. Radiographics 24(5):1317–1330
Kerrison C, Davidson JE, Cleary AG et al (2004) Pamidronate in the treatment of childhood SAPHO syndrome. Rheumatology (Oxford) 43(10):1246–1251
Khanna G, Sato TS, Ferguson P (2009) Imaging of chronic recurrent multifocal osteomyelitis. Radiographics 29(4):1159–1177
King SM, Laxer RM, Manson D et al (1987) Chronic recurrent multifocal osteomyelitis: a noninfectious inflammatory process. Pediatr Infect Dis J 6(10):907–911
Land C, Rauch F, Munns CF et al (2006) Vertebral morphometry in children and adolescents with osteogenesis imperfecta: effect of intravenous pamidronate treatment. Bone 39(4):901–906
Maksymowych WP (2003) Bisphosphonates for arthritis–a confusing rationale. J Rheumatol 30(3):430–434
Maksymowych WP, Lambert R, Jhangri GS et al (2001) Clinical and radiological amelioration of refractory peripheral spondyloarthritis by pulse intravenous pamidronate therapy. J Rheumatol 28(1):144–155
Miettunen PM, Wei X, Kaura D et al (2009) 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 7:2
Munns CF, Rajab MH, Hong J et al (2007) Acute phase response and mineral status following low dose intravenous zoledronic acid in children. Bone 41(3):366–370
Munns CF, Rauch F, Travers R et al (2005) Effects of intravenous pamidronate treatment in infants with osteogenesis imperfecta: clinical and histomorphometric outcome. J Bone Miner Res 20(7):1235–1243
Olivieri I, Padula A, Ciancio G et al (2002) Successful treatment of SAPHO syndrome with infliximab: report of two cases. Ann Rheum Dis 61(4):375–376
Olivieri I, Padula A, Ciancio G et al (2003) Persistent efficacy of tumor necrosis factor alpha blockage therapy in SAPHO syndrome: comment on the article by Wagner et al. Arthritis Rheum 48(5):1467, author reply 1467-8
Rosenberg ZS, Shankman S, Klein M et al (1988) Chronic recurrent multifocal osteomyelitis. AJR Am J Roentgenol 151(1):142–144
Schilling F, Fedlmeier M, Eckardt A et al (2002) Vertebral manifestation of chronic recurrent multifocal osteomyelitis (CRMO). Rofo 174(10):1236–1242
Simm PJ, Allen RC, Zacharin MR (2008) Bisphosphonate treatment in chronic recurrent multifocal osteomyelitis. J Pediatr 152(4):571–575
Toussirot E, Wendling D (2007) Antiinflammatory treatment with bisphosphonates in ankylosing spondylitis. Curr Opin Rheumatol 19(4):340–345
Vanhoenacker FM, Baekelandt J, Vanwambeke K et al (1998) Chronic recurrent multifocal osteomyelitis. J Belge Radiol 81(2):84–86
Wagner AD, Andresen J, Jendro MC et al (2002) Sustained response to tumor necrosis factor alpha-blocking agents in two patients with SAPHO syndrome. Arthritis Rheum 46(7):1965–1968
Yu L, Kasser JR, O’Rourke E et al (1989) Chronic recurrent multifocal osteomyelitis. Association with vertebra plana. J Bone Joint Surg Am 71(1):105–112
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We would like to thank Dr. Peter Ainslie for linguistic assistance.
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Hospach, T., Langendoerfer, M., von Kalle, T. et al. Spinal involvement in chronic recurrent multifocal osteomyelitis (CRMO) in childhood and effect of pamidronate. Eur J Pediatr 169, 1105–1111 (2010). https://doi.org/10.1007/s00431-010-1188-5
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DOI: https://doi.org/10.1007/s00431-010-1188-5