Chronic recurrent multifocal osteomyelitis in children: a single center experience over five years

Turk J Pediatr. 2019;61(3):386-391. doi: 10.24953/turkjped.2019.03.010.

Abstract

Sağ E, Sönmez HE, Demir S, Bilginer Y, Ergen FB, Aydıngöz Ü, Özen S. Chronic recurrent multifocal osteomyelitis in children: a single center experience over five years. Turk J Pediatr 2019; 61: 386-391. Chronic recurrent multifocal osteomyelitis (CRMO) is a rare disease characterized by sterile bone inflammation. It is an orphan disease with many unclear aspects in terms of diagnosis, treatment and follow-up. The aim of this study was to report our experience of pediatric CRMO patients. Children who were diagnosed with CRMO, and were followed-up between January 2008 and January 2017, were included in this study. There were 15 CRMO patients (8M/7F) with a median age at diagnosis of 9.0 years (range: 0.6-15.0). Bone pain was the most common presenting symptom. All of the patients had multifocal bone lesions. Vertebrae (66.7%) and femur (66.7%) were the most commonly affected bones. Eight of the patients also had sacroiliitis; however, only one of them was HLA-B27 positive. Whole-body magnetic resonance imaging (MRI) was used as a diagnostic tool in 13 patients revealing bone marrow edema (84.6%), osteitis (69.2%), and periosteal reaction (61.5%). All patients were initially treated with non-steroidal anti-inflammatory drugs (NSAIDs), however, disease-modifying anti-rheumatic drugs, anti-TNF agents or pamidronate were added to therapy due to inadequate treatment response. Clinical remission was achieved in 12 patients (1 with NSAIDs, 3 with methotrexate, 1 with pamidronate and 7 with an anti-TNF agent). During the follow-up period, relapses were observed in four patients who presented with pain and/or a newly formed bone lesion on MRI. Eventually, however, all of these patients also reached remission. CRMO is a chronic disease which may have a progressive or relapsing-remitting course. Improvement of the knowledge about this rare disease may help to enlighten the unknowns of the disease.

Keywords: CRMO; chronic recurrent multifocal osteomyelitis; treatment.

MeSH terms

  • Adolescent
  • Anti-Inflammatory Agents, Non-Steroidal / therapeutic use
  • Antirheumatic Agents / therapeutic use
  • Blood Sedimentation
  • Bone Marrow Diseases / diagnostic imaging
  • C-Reactive Protein / analysis
  • Child
  • Child, Preschool
  • Edema / diagnostic imaging
  • Female
  • Follow-Up Studies
  • Humans
  • Infant
  • Magnetic Resonance Imaging
  • Male
  • Osteitis / diagnostic imaging
  • Osteomyelitis / diagnosis*
  • Osteomyelitis / drug therapy*
  • Periosteum / diagnostic imaging
  • Recurrence
  • Tumor Necrosis Factor-alpha / antagonists & inhibitors
  • Whole Body Imaging

Substances

  • Anti-Inflammatory Agents, Non-Steroidal
  • Antirheumatic Agents
  • Tumor Necrosis Factor-alpha
  • C-Reactive Protein

Supplementary concepts

  • Chronic recurrent multifocal osteomyelitis