Elsevier

Joint Bone Spine

Volume 68, Issue 3, May 2001, Pages 245-251
Joint Bone Spine

ORIGINAL ARTICLE
Chronic recurrent multifocal osteomyelitis: five-year outcomes in 14 pediatric cases

https://doi.org/10.1016/S1297-319X(01)00276-7Get rights and content

Abstract

Objective. To determine the clinical presentation and outcomes of chronic recurrent multifocal osteomyelitis (CRMO) in pediatric patients. Patients and methods. Ten girls and four boys were followed up between 1993 and 1999 for CRMO diagnosed on the basis of radiographic bone lesions with, at the same sites, increased radionuclide uptake, negative microbiological specimens, and histological evidence of nonspecific osteomyelitis. Results. Mean age at CRMO was 9.6 ± 3.4 years, mean disease duration was 5.3 ± 2.5 years, and mean number of flares per patient was 5.9 ± 3.7. Thirty-four percent of lesions were in the metaphyses of the lower limb bones, 14% in the pelvis, and 13% in the chest wall (with clavicular lesions in four patients). Three patients had skin lesions (psoriasis in two and palmoplantar pustulosis in one). Eight patients received antibiotic therapy, for 2 months at the most, to no advantage in the short term. Nonsteroidal anti-inflammatory drugs were used in all 14 patients and glucocorticoid therapy in four. Sulfasalazine was used in five patients, to good effect in four. Mean follow-up was 5.3 ± 2.5 years. At last follow-up, eight patients had active disease, including one with synovitis and one with Takayashu’s disease. Conclusion. As compared to SAPHO syndrome, skin lesions and chest wall involvement are less common in CRMO. The long-term prognosis is guarded: in our study only six of 14 patients were in remission at last follow-up.

Section snippets

Patients and methods

We retrospectively reviewed the medical charts of all the pediatric patients who received follow-up for CRMO at the pediatric rheumatology department of the Saint Vincent de Paul and Robert Debré teaching hospitals in Paris, France between 1993 and 1999. The diagnosis was based on presence of suggestive radiographic bone lesions with increased radionuclide uptake on bone scans and evidence of noninfectious osteomyelitis by biopsy. The following data were abstracted from the charts: age and sex;

Patient characteristics and clinical presentation

Fourteen patients met our criteria for CRMO, ten girls and four boys, with a mean age of 9.6 ± 3.4 years at disease onset. Psoriasis developed in two patients, 6 and 7 years after the first bone symptoms, respectively. Another patient had palmoplantar pustulosis coincident with the first bone symptoms. One patient had a family history of psoriasis, two with a history of ill-defined musculoskeletal disease, and one of ankylosing spondylitis. Inflammatory pain was present at onset in all 14

Discussion

Our findings are in keeping with those of earlier series showing a female predominance of about 70% and a mean age at onset of 9 to 10 years 3, 4, 5. Palmoplantar pustulosis and psoriasis seem less common in children with CRMO than in adults with SAPHO syndrome syndrome 〚30〛; furthermore, the skin lesions can occur many years after the first bone symptoms 〚6〛. Of the 22 patients studied by Carr et al. 〚7〛, only two had palmoplantar pustulosis, and one psoriasis, whereas pustulosis was present

Conclusion

As compared to SAPHO syndrome, CRMO is less likely to cause skin lesions and anterior chest wall involvement. Flares occur at variable intervals, and the disease can remain active into adulthood. Nonsteroidal anti-inflammatory drugs are the treatment of choice, as they are usually both effective and well tolerated. In severe forms refractory to nonsteroidal anti-inflammatory drugs, glucocorticoid therapy is required. However, the adverse effects of glucocorticoids, particularly on growth, are

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