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Biologic treatment for chronic recurrent multifocal osteomyelitis: report of four cases and review of the literature

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Abstract

Chronic recurrent multifocal osteomyelitis (CRMO) is a rare non-infectious inflammatory disorder with unpredictable clinical course, characterized by acute exacerbations and spontaneous remissions. There are no randomized-controlled trials about treatment options. Non-steroidal anti-inflammatory drugs (NSAID) are the first-line treatment option; glucocorticoids seem to be effective; positive outcomes have been obtained with bisphosphonates. In the last few years successful use of biologic agents like anti-TNF agents has been reported. We report the cases of 3 children suffering from CRMO who were treated with NSAID, steroid, bisphosphonates and eventually received etanercept and 1 case without vertebral involvement treated with etanercept after NSAID and steroid; all cases showed clinical improvement. The mean ages at symptoms onset and diagnosis were 8 and 10 years and 10 months, respectively. Two patients presented with back pain and three had vertebral lesions. Mean interval from diagnosis to the onset of anti-TNF treatment was 14 months. According to our small experience, we suggest considering therapy with etanercept for the treatment of severe cases with persistently active disease despite multiple treatments.

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Authors and Affiliations

  1. Pediatric Unit, Department of Medical and Surgical Sciences, S. Orsola-Malpighi Hospital, University of Bologna, Via Massarenti 9, Bologna, Italy

    Elena Tronconi, Angela Miniaci & Andrea Pession

  2. Pediatric Radiology Unit, Sant’Orsola-Malpighi Hospital, University of Bologna, Bologna, Italy

    Michelangelo Baldazzi & Laura Greco

Authors
  1. Elena Tronconi
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  2. Angela Miniaci
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  3. Michelangelo Baldazzi
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  4. Laura Greco
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  5. Andrea Pession
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Contributions

Authors disclose involvement of any external agent in language editing. ET and AM conceptualized and designed the study; acquired, analysed and interpreted the data, drafted the initial manuscript, and approved the final manuscript as submitted. MB and LG performed the radiological exams, acquired, analysed and interpreted the data, critically reviewed the manuscript, and approved the final manuscript as submitted. AP substantial contributed to the conception of the study, revised the manuscript critically for important intellectual content, and gave final approval of the version to be published.

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Correspondence to Elena Tronconi.

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The authors declare that they have no conflict of interest.

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This article does not contain any studies with human participants or animals performed by any of the authors. Standard care treatment was provided, and the patients were not part of a research study. For this type of article, formal ethical approval is not required.

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Consent from the parents/LARs of the patients below 16 years for publication was obtained.

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Tronconi, E., Miniaci, A., Baldazzi, M. et al. Biologic treatment for chronic recurrent multifocal osteomyelitis: report of four cases and review of the literature. Rheumatol Int 38, 153–160 (2018). https://doi.org/10.1007/s00296-017-3877-0

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  • DOI: https://doi.org/10.1007/s00296-017-3877-0

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