Evolution of chronic recurrent multifocal osteitis toward spondylarthropathy over the long term

Abstract

Objective. To retrospectively assess, with a sufficiently long followup (mean 11.6 years; median 9 years), the long-term outcome of chronic recurrent multifocal osteitis (CRMO), a multifocal, inflammatory bone disease.Methods. Patients included were 8 children/ adolescents and 7 adults with no family history of rheumatic disease who had been diagnosed as having CRMO between 1979 and 1995. Ten patients had undergone at least 1 bone biopsy of the lesions, with histologic examination and multiple cultures. In 1996, in addition to an in-depth interview, 12 patients underwent an extensive physical examination, laboratory evaluation, HLA-A, B, C, and DR typing, bone radiography and scintigraphy, and computed tomography scan of the sternoclavicular and sacroiliac joints.Results. Remission was observed in 3 patients. The other 12 patients developed various associations of vertebral (n ‫؍‬ 10), sacroiliac (n ‫؍‬ 6), anterior thoracic (n ‫؍‬ 7), peripheral articular (n ‫؍‬ 2), enthesopathic (n ‫؍‬ 4), or dermatologic (palmoplantar pustulosis in 3 cases and psoriasis in 2) involvements. Spine involvement was the most common and occurred the earliest (median time to appearance after the onset of osteitis 5.63 years). Clinical sacroiliitis was always unilateral. No patients carried the HLA-B27 haplotype. CRMO responded well to nonsteroidal antiinflammatory drugs. Twelve patients met the European Spondylarthropathy Study Group criteria for spondylarthopathy.

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

“…9 These fi ndings are similar to the clinical, radiological, and pathological fi ndings commonly associated with sternocostoclavicular hyperostosis or palmoplantar pustulosis-related arthritis. 10 We thus believe that these diseases belong to the same disease category as CRMO. The present patient demonstrated a single chronic infl ammatory lesion in the marrow of her right femoral Inflammatory granulation osteoblast osteoclast Fig.…”

Chronic Recurrent Multifocal Osteomyelitis

“…The duration of active disease has been linked to the initial and total number of bone lesions and inversely correlated with the age of onset [10,40]. It has also been described that CRMO can evolve into a spondyloarthropathy, with clinical and radiological sacroiliitis [19]. Enthesitis related arthritis and psoriatic arthritis have been reported in the long-term follow-up of several cohorts of children and adolescents with CRMO, despite being treated with anti-inflammatory drugs [9,11,15,19].…”

A missense mutation in pstpip2 is associated with the murine autoinflammatory disorder chronic multifocal osteomyelitis

“…It presents in childhood with multiple, painful, sterile, inflammatory bone lesions, often accompanied by fever [1,2]. It is frequently seen in association with other inflammatory disorders including psoriasis and inflammatory bowel disease [3][4][5][6][7][8][9].…”