Elsevier

Human Pathology

Volume 30, Issue 1, January 1999, Pages 59-65
Human Pathology

Original contribution
Chronic recurrent multifocal osteomyelitis in children: Diagnostic value of histopathology and microbial testing

https://doi.org/10.1016/S0046-8177(99)90301-5Get rights and content

Abstract

Chronic recurrent, unifocal or multifocal osteomyelitis (CRMO), an inflammatory disorder of unknown origin, involves different osseous sites and may be associated with palmoplantar pustulosis. Bacterial cultures of affected tissue were reported negative in nearly all cases. Radiological and magnetic resonance imaging features of CRMO have been described, but differential diagnosis remains difficult, including rheumatic diseases, bacterial osteomyelitis, and malignancy. Although definite diagnosis relies on histopathologic confirmation by biopsy, histopathologic criteria have not been defined. Because CRMO may be treated with nonsteroidal antiinflammatory drugs, but not antibiotics, distinguishing CRMO from bacterial osteomyelitis is of major importance. Histopathologic analysis of 12 patients with CRMO indicated a wide variation of reparative changes of bone, but chronic inflammation could not be found at all sites in the same biopsy. The inflammatory infiltrate was mostly scattered, consisting mainly of lymphocytes, plasma cells, histiocytes, and also few neutrophil granulocytes. Immunohistochemistry showed a predominance of CD3(+), CD45RO(+) T-cells, which were mainly CD8(+). In addition, CD20(+) B cells and CD68(+) macrophages were abundant in each biopsy specimen. Mild lymphocytic and granulocytec infiltrates were also detected in three synovial biopsy specimens obtained from adjacent joints. All bacterial and fungal cultures from native biopsy tissues were negative. Amplification of partial-length 16S ribosomal DNA by polymerase chain reaction (PCR) using broad-range eubacterial primers was below the detection limit in all patients. Because histopathologic features alone may not provide conclusive evidence, CRMO should be included in the differential diagnosis of chronic inflammatory bone lesions in children, and the definite diagnosis should be made by the clinical picture, x-ray studies, bone scan, bacterial culture, and histopathologic analysis in a multidisciplinary approach.

References (35)

  • D Harmsen et al.

    Heterogeneity among Whipple's-disease-associated bacteria

    Lancet

    (1994)
  • B Björksten et al.

    Chronic recurrent multifocal osteomyelitis and pustulosis palmoplantaris

    J Pediatr

    (1978)
  • A Giedion et al.

    Subacute and chronic “symmetrical” osteomyelitis

    Ann Radiol Paris

    (1972)
  • AJ Carr et al.

    Chronic multifocal osteomyelitis

    J Bone Joint Surg Br

    (1993)
  • EO Gerscovich et al.

    Osteomyelitis of the clavicle: Clinical and bacteriological findings in ten patients

    Skeletal Radiol

    (1994)
  • A Mortensson et al.

    Chronic recurrent multifocal osteomyelitis in children: A roentgenologic and scintigraphic investigation

    Acta Radiol

    (1988)
  • RS van Howe et al.

    Chronic, recurrent multifocal osteomyelitis

    Clin Pediatr

    (1989)
  • HJ Girschick et al.

    Chronic recurrent osteomyelitis with clavicular involvement in children: Diagnostic value of different imaging techniques and therapy with nonsteroidal anti-inflammatory drugs

    Eur J Pediatr

    (1998)
  • Case report of the Massachusetts General Hospital

    N Engl J Med

    (1986)
  • AG Jurik et al.

    Chronic sclerosing osteomyelitis of the clavicle: A manifestation of chronic recurrent multifocal osteomyelitis

    Arch Orthop Trauma Surg

    (1987)
  • RG Appell et al.

    Condensing osteitis of the clavicle in childhood: A rare sclerotic bone lesion

    Pediatr Radiol

    (1983)
  • M Lissens et al.

    Condensing osteitis of the clavicle: Report of two cases and review of the literature

    Medica Physica

    (1990)
  • JP Huaux et al.

    Pustulotic arthroosteitis and chronic recurrent multifocal osteomyelitis in children: Report of three cases

    J Rheumatol

    (1988)
  • MF Kahn et al.

    Sternoclavicular pustulotic osteitis (SAPHO): 20-year interval between skin and bone lesions

    J Rheumatol

    (1991)
  • MF Kahn

    Psoriatic arthritis and synovitis, acne, pustulosis, hyperostosis and osteitis syndrome

    Curr Opin Rheumatol

    (1993)
  • CL Benhamou et al.

    Synovitis-acnepustulosis hyperostosis-osteomyelitis syndrome (SAPHO): A new syndrome among the spondyloarthropathies?

    Clin Exp Rheum

    (1988)
  • B Björksten et al.

    Histopathological aspects of chronic recurrent multifocal osteomyelitis

    J Bone Joint Surg Br

    (1980)
  • Cited by (0)

    View full text