Chronic recurrent multifocal osteomyelitis (CRMO) in childhood – review and original results

Czech version

Authors: K. Bouchalova ^1,2; J. Fráňová ²; M. Schüller ²; J. Skotáková ³; M. Macků ²
Authors‘ workplace: Dětská klinika, Ústav molekulární a translační medicíny LF UP a FN, Olomouc ¹; Pediatrická klinika LF MU a FN, Brno ²; Klinika dětské radiologie LF MU a FN, Brno ³
Published in: Čes. Revmatol., 27, 2019, No. 3, p. 116-124.
Category:

Overview

Aim was both detailed review of chronic recurrent multifocal osteomyelitis (CRMO) at first part of paper, and retrospective analysis of our pediatric patients with focus on diagnostics and therapy. Specific aim was analysis of pamidronate therapy.

Methods. Data on 20 children with CRMO (15 female and 5 male, ratio 3 : 1) treated at Pediatric Department of the University Hospital Brno between January 2011 – May 2016 were analyzed, including epidemiologic, clinical, laboratory, and radiology data at the time of diagnosis and during follow-up.

Results. Symptomatic sites were predominantly lower extremities (all patients), followed by clavicle (35% patients), spine (35%), upper extremities (35%), and sternum (10%). Whole body MRI and biopsy were performed in 7/20 (35%) and 14/20 (70%) patients, respectively. At least one laboratory inflammatory marker was elevated in 15 patients, but CRP elevation above 30 mg/L was found only in 4 children. Median follow-up from time of diagnosis was 3.6 years (range 0.2 to 8.8 years). Prednisone (in 6 children) and methotrexate (MTX in 4 children) had not been effective in terms of the long term outcome. On the other hand, intravenous pamidronate and etanercept showed good efficacy. Pamidronate was used in 14/20 (70%) children. In 13/14 (93%) out of them inactive disease or improvement of status was present. In 9/14 (64%) patients termination of pamidronate was performed due to sustained clinical inactivity (absence of pain, local and systemic signs of inflammation, including laboratory inflammatory markers). Etanercept was successfully used in two patients.

Conclusion. In case of nonsteroidal antiinflammatory drugs monotherapy failure pamidronate therapy showed excellent results, without serious side effects. On the other hand, corticosteroids and MTX had only short effect. Further research should be focused on markers of remission, and a pamidronate regimen optimization.

Keywords:

chronic recurrent multifocal osteomyelitis – whole body magnetic resonance imaging – pamidronate

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Czech Rheumatology

2019 Issue 3