Chronic recurrent multifocal osteomyelitis in children

Schultz, Christian; Holterhus, Paul Martin; Seidel, A.; Jonas, S.; Barthel, M.; Kruse, K.; Bucsky, Peter

doi:10.1097/00006454-199911000-00015

Cited by 159 publications

(154 citation statements)

References 38 publications

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“…This is consistent with the findings of earlier studies that investigated the distribution of patient characteristics in CNO cases (23,24). There was no evidence that one gender was affected with CNO at an earlier age than the other.…”

Section: Discussionsupporting

confidence: 91%

“…There was no evidence that one gender was affected with CNO at an earlier age than the other. The median number of bone lesions per patient was 3.6, similar to that reported previously (15,24). With regard to the number of affected bones per patient, we cannot exclude that affected bones could have been overlooked for several reasons.…”

Section: Discussionsupporting

confidence: 83%

“…With 60 bone lesions identified, the lower extremity was the most affected part of the skeleton, similar to the findings of previous studies (15,16,26). A previous study showed an association between CNO and different skin manifestations, such as palmoplantar pustulosis and acne, in up to 23% of children (24). In our study, we found an association rate of 21%.…”

Section: Discussionsupporting

confidence: 72%

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Characteristics and outcomes of chronic non-bacterial osteitis in children

Wintrich¹,

Horneff²

2015

Eur J Rheumatol

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Objective: To determine patient characteristics, clinical presentation, pattern of involvement, treatment, and outcome of patients with chronic non-bacterial osteitis (CNO).Material and Methods: Consecutive cases of CNO were analyzed at a single center for pediatrics and adolescent medicine from 2006 to 2013 in terms of patient characteristics, clinical presentation, pattern of involvement, treatment, and outcome. Results:We identified 32 children aged 1.5-15 years who were diagnosed with CNO between 2006 and 2013. A maximum of 12 bones per patient were affected in a total of 114 documented locations. The pelvis and clavicle (affecting 34% of patients each) were the most frequently affected bones. The foot skeleton was the most commonly affected region in 60% of patients. Skin manifestations were found in 7 (21%) patient. Increased inflammatory signs at presentation were detected in 18 patients. Pathological findings were found in all 30 children examined using magnetic resonance imaging (MRI), in 10 of 11 children examined using radiography, and in 8 of 10 patients examined using skeletal scintigraphy. Bone biopsy was performed in 9 patients. For initial treatment, non-steroidal anti-inflammatory drugs (NSAIDs) or coxibs were used in 28 (87.5%) patients. Remission or satisfactory follow-up was achieved in all patients.Conclusion: Today, CNO is increasingly diagnosed using MRI and rarely through histological examinations. Therapeutic strategies include NSAIDs, which are often highly effective. All patients in the present study showed good clinical outcomes. Keywords: Chronic recurrent multifocal osteomyelitis, bone diseases, chronic disease IntroductionChronic non-bacterial osteitis (CNO) is a benign noninfectious autoinflammatory disease of the bone tissue with an incomplete etiology (1, 2). The incidence of CNO is 0.4/100.000 children (3). It was first reported by Giedion et al. (4) in 1972. There are several synonyms for CNO, such as chronic recurrent multifocal osteomyelitis (CRMO); non-bacterial osteitis; or synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome, all describing the same disease entity (5, 6). CNO generally occurs in children and adolescents, with a peak age of onset of 7-12 years (7). Localized bone pain is the leading symptom, often with local swelling and warmth (8). Patients are usually in good general condition. CNO can appear as a mono-/oligo-focal disease, as well as in chronic recurrent polyfocal stages with a risk of late effects, such as vertebral fractures and severe hyperostotic bone lesions (9). CNO primarily affects the metaphyses of long bones, although lesions can occur in any part of the skeleton (10). Other organs, including the skin, eyes, gastrointestinal tract, and lungs, can also be affected by inflammation (9,11,12). Skin inflammation manifests as palmoplantar pustulosis, acne, psoriasis, and rarely as pyoderma gangrenosum (1, 2). The most important differential diagnosis is bacterial osteomyelitis, although it is not usually multifocal (13). Even...

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Section: Discussionsupporting

confidence: 91%

Section: Discussionsupporting

confidence: 83%

Section: Discussionsupporting

confidence: 72%

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Characteristics and outcomes of chronic non-bacterial osteitis in children

Wintrich¹,

Horneff²

2015

Eur J Rheumatol

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“…Upon whole-body imaging, other asymptomatic sites of disease can often be identified. CRMO is also associated with the presence of cutaneous lesions, with a prevalence about 25 % in children [10]. The differential diagnosis includes subacute and chronic infectious osteomyelitis, histiocytosis, hypophosphatasia, and infiltrative malignancies such as leukemia, lymphoma, and Ewing's sarcoma in children [11].…”

Section: Discussionmentioning

confidence: 99%

“…Nonsteroidal anti-inflammatory drugs are first-line therapy and usually effective with a response rate up to 80 % [10]. Other possible pharmacologic treatments include bisphosphonates, sulfasalazine, methotrexate, colchicine, interferons, and gamma globulins [23].…”

Section: Discussionmentioning

confidence: 99%

Chronic recurrent multifocal osteomyelitis with an atypical presentation in an adult man

et al. 2015

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We present the case of a 33-year-old man with no significant medical history who developed right scapular pain, left-sided sacroiliac joint pain, and lower back pain, and was eventually diagnosed with chronic recurrent multifocal osteomyelitis (CRMO). Imaging demonstrated multiple scattered T2-hyperintense lesions on MRI at the spine and the left SI joint, some of which progressed and one regressed in size on follow-up. Histopathology demonstrated only non-specific chronic inflammation compatible with CRMO. No evidence of infectious organisms or neoplastic processes was found. The pain was relapsing and remitting in nature. Laboratory investigations were notable for no evidence of hematologic malignancy or infection, but only a mild increase in alkaline phosphatase. This case highlights that CRMO, despite being thought of as a childhood-onset disease, can present in adults as well, and also provides illustrative examples of imaging and histological findings.

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