Abstract
Background
Chronic recurrent multifocal osteomyelitis (CRMO) is an autoinflammatory disorder that is currently diagnosed based on clinical, radiologic, pathological and longitudinal findings.
Objective
To provide detailed descriptions of CRMO lesion patterns seen on radiographs and MRI and to suggest clinical use of whole-body MRI and propose noninvasive diagnostic strategy.
Materials and methods
Retrospective longitudinal study (1989–2010) of 31 children (22 girls, 9 boys) diagnosed with CRMO. Imaging data were evaluated by two pediatric radiologists.
Results
Mean age at diagnosis was 11 years (3–17). A total of 108 lesions were investigated. The most common sites were the long bone metaphyses (56 lesions in 24 children) especially femoral and tibial (20/24); pelvis (10/31); spine (9/31); clavicle (6/31) and mandible (3/31). In long bones, the radiologic appearance was normal (22/56), mixed lytic and sclerotic (20/56), sclerotic (8/56) or lytic (6/56) often juxtaphyseal (36/56), with hyperostosis or periosteal thickening (10/56). Vertebral involvement was often multifocal (6/9). Medullary edema was seen on MRI (42) with epiphyseal (23/42) or soft-tissue (22/42) inflammation and juxtaphyseal nodule-like appearance (7/42). Whole-body MRI (15/31) was key in detecting subclinical lesions.
Conclusion
CRMO is a polymorphous disorder in which whole-body MRI is extremely useful for showing subclinical edema. Vertebral collapse requires long-term monitoring.
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References
Giedion A, Holthusen W, Masel LF et al (1972) Subacute and chronic symmetrical osteomyelitis. Ann Radiol 15:329–342
Probst FP (1976) Chronic multifocal cleido-metaphyseal osteomyelitis in childhood. Report of a case. Acta Radiol Diagn 17:531–537
Demharter J, Bohndorf K, Michl W et al (1997) Chronic recurrent multifical osteomyelitis: a radiological and clinical investigation of five cases. Skeletal Radiol 26:579–588
Brown T, Wilkinson RH (1988) Chronic recurrent multifocal osteomyelitis. Radiology 166:493–496
King SM, Laxer RM, Manson D et al (1987) Chronic recurrent multifocal osteomyelitis: a noninfectious inflammatory process. Pediatr Infect Dis J 6:907–911
Handrick W, Hörmann D, Voppmann A et al (1998) Chronic recurrent multifocal osteomyelitis. Report of eight children. Pediatr Surg Int 14:195–198
Iyer RS, Thapa MM, Chew FS (2011) Chronic recurrent multifocal osteomyelitis: review. AJR Am J Roentgenol 196:S87–S91
Gikas PD, Islam L, Aston W et al (2009) Nonbacterial osteitis: a clinical, histopathological, and imaging study with a proposal for protocol-based management of children with this diagnosis. J Orthop Sci 14:505–516
Mortensson W, Edeburn G, Fries M et al (1988) Chronic recurrent multifocal osteomyelitis in children. A roentgenologic and scintigraphic investigation. Acta Radiol 29:565–570
Cyrlak D, Pais MJ (1986) Chronic recurrent multifocal osteomyelitis. Skeletal Radiol 15:32–39
Yu L, Kasser JR, O’Rourke E et al (1989) Chronic recurrent multifocal osteomyelitis. Association with vertebra plana. J Bone Joint Surg Am 71:105–112
Jansson A, Renner ED, Ramser J et al (2007) Classification of non-bacterial osteitis: retrospective study of clinical, immunological and genetic aspects in 89 children. Rheumatology 46:154–160
Khanna G, Sato TS, Ferguson P (2009) Imaging of chronic multifocal osteomyelitis. Radiographics 29:1159–1177
Jurriaans E, Singh NP, Finlay K et al (2001) Imaging of chronic recurrent multifocal osteomyelitis. Radiol Clin North Am 39:305–327
Jurik AG, Egund N (1997) MRI in chronic recurrent multifocal osteomyelitis. Skeletal Radiol 26:230–238
Manson D, Wilmot DM, King S et al (1989) Physeal involvement in chronic recurrent multifocal osteomyelitis. Pediatr Radiol 20:76–79
Hospach T, Langendoerfer M, von Kalle T et al (2010) Spinal involvement in chronic recurrent multifocal osteomyelitis (CRMO) in childhood and effect of pamidronate. Eur J Pediatr 169:1105–1111
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 32:328–336
Fritz J, Tzaribatchev N, Claussen CD et al (2009) Chronic recurrent multifical osteomyelitis: comparison of whole-body MR imaging with radiography and correlation with clinical and laboratory data. Radiology 252:842–851
Guérin-Pfyffer S, Guillaume-Czitrom S, Tammam S et al (2012) Evaluation of chronic recurrent multifocal osteitis in children by whole-body magnetic resonance imaging. J Bone Spine [epub ahead of print]
Stedman TL (2003) Stedman’s medical dictionary, 27th edn. Lippincot Williams & Wilkins, Philadelphia
Girschick HJ, Raab P, Surbaum S et al (2005) Chronic nonbacterial osteomyelitis in children. Ann Rheum Dis 64:279–285
Catalano-Pons C, Comte A, Wipff J et al (2008) Clinical outcome in children with chronic recurrent multifocal osteomylitis. Rheumatology 47:1397–1399
Huber AH, Lam PY, Duffy CM et al (2002) Chronic recurrent multifocal osteomyelitis: clinical outcomes after more than five years of follow-up. J Pediatr 141:198–203
Job-Deslandre C, Krebs S, Kahan A (2001) Chronic recurrent multifocal osteomyelitis: five-year outcomes in 14 pediatric cases. Joint Bone Spine 68:245–251
Gustavson KH, Wilbrand HF (1974) Chronic symmetric osteomelitis. Report of a case. Acta Radiol 15:551–557
Probst FP, Bjorkstein B, Gustavson KH (1978) Radiological aspect of chronic recurrent multifocal osteomyelitis. Ann Radiol 21:115–125
Björkstén B, Boquist L (1980) Histopathological aspects of chronic recurrent multifocal osteomyelitis. J Bone Joint Surg 62:376–380
Earwaker JWS, Cotten A (2003) SAPHO: syndrome or concept? Imaging findings. Skeletal Radiol 32:311–327
Beretta-Piccoli BC, Sauvain MJ, Gal I et al (2000) Synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome in childhood: a report of ten cases and review of the literature. Eur J Pediatr 159:594–601
El Shanti H, Ferguson PJ (2007) Chronic recurrent multifocal osteomyelitis: a concise review and gentic update. Clin Orthop Relat Res 462:11–19
Ferguson PJ, Bing X, Vaself MA et al (2006) A missense mutation in pstpip2 is associated with the murine autoinflammatory disorder chronic multifocal osteomyelitis. Bone 38:41–47
Grosse J, Chitu V, Marquardt A et al (2006) Mutation of mouse Mayp/Pstpip2 causes a macrophage autoinflammatory disease. Blood 107:3350–3358
Quelquejay C, Job-Deslandre C, Hamidou A et al (1997) [Chronic recurrent multifocal osteomyelitis in children]. J Radiol 78:115–121
Mandell GA, Contreras SJ, Conard K et al (1998) Bone scintigraphy in the detection of chronic recurrent multifocal osteomyelitis. J Nucl Med 39:1778–1783
Chow LT, Griffith JF, Kumta SM et al (1999) Chronic recurrent multifocal osteomyelitis: a great clinical and radiological mimic in need of recognition by the pathologist. APMIS 107:369–379
Pöyhiä T, Azouz EM (2000) MR imaging evaluation of subacute and chronic bone abcesses in children. Pediatr Radiol 30:763–768
Wang A, Babyn P (2008) Joint lesions in children with chronic recurrent multifocal osteomyelitis. RSNA. http://rsna2008.rsna.org/event_display.cfm?em_id=6005684. Abstract
Sundaram M, McDonald D, Engel E et al (1996) Chronic recurrent multifocal osteomyelitis: an evolving clinical and radiological spectrum. Skeletal Radiol 25:333–336
Jaramillo D (2010) Whole-body MR imaging, bone diffusion imaging: how and why? Pediatr Radiol 40:978–984
Baur-Melnyk A (2009) Malignant versus benign vertebral collapse: are new imaging techniques useful? Cancer Imaging 9:S49–S51
Girschik HJ, Krauspe R, Tschammler A et al (1998) Chronic recurrent osteomyelitis with clavicular involvement in children: diagnosis value of different imaging techniques and therapy with non-steroidal anti-inflammatory drugs. Eur J Pediatr 157:28–33
Appell RG, Oppermann HC, Becker W et al (1983) Condensing osteitis of the clavicle in childhood: a rare sclerotic bone lesion. Review of literature and report of seven children. Pediatr Radiol 13:301–306
Kahn MF, Hayem F, Hayem G et al (1994) Is diffuse scleroting osteomyelitis of the mandible part of the synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome? Oral Surg Oral Med Oral Pathol 78:594–598
Suei Y, Taguchi A, Tanimoto K (1996) Diffuse sclerosing osteomyelitis of the mandible: its characteristics and possible relationship to synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome. J Oral Maxillofac Surg 54:1194–1199, discussion 1199–1200
Darge K, Jaramillo D, Siegel MJ (2008) Whole-body MRI in children current status and future applications. Eur J Radiol 68:289–298
Goo HW, Yang DH, Ra YS et al (2006) Whole-body MRI of Langerhans cell histiocytosis: comparison with radiography and bone scintigraphy. Pediatr Radiol 36:1019–1031
Chavhan GB, Babyn PS (2011) Whole-body MR imaging in children: principles, technique, current applications, and future directions. Radiographics 31:1757–1772
Martin JC, Desoysa R, O’Sullivan MM et al (1996) Chronic recurrent multifocal osteomyelitis: spinal involvement and radiological appearances. Br J Rheumatol 35:1019–1021
Ording Müller LS, Avenarius D (2011) High signal in bone marrow at diffusion-weighted imaging with body background suppression (DWIBS) in healthy children. Pediatr Radiol 41:221–226
Duffy CM, Lam PY, Ditchfield M et al (2002) Chronic recurrent multifocal osteomyelitis: review of orthopaedic complications at maturity. J Pediatr Orthop 22:501–505
Baulot E, Bouillien D, Giroux EA et al (1998) Chronic recurrent multifocal osteomyelitis causing spinal cord compression. Eur Spine J 7:340–343
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The authors thank Pascale Zerbini for manuscript preparation.
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Falip, C., Alison, M., Boutry, N. et al. Chronic recurrent multifocal osteomyelitis (CRMO): a longitudinal case series review. Pediatr Radiol 43, 355–375 (2013). https://doi.org/10.1007/s00247-012-2544-6
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DOI: https://doi.org/10.1007/s00247-012-2544-6