Clinical experiences in Turkish paediatric patients with chronic recurrent multifocal osteomyelitis

Turk J Pediatr. 2019;61(6):879-884. doi: 10.24953/turkjped.2019.06.009.

Abstract

Sözeri B, Aktay Ayaz N, Yıldız Atıkan B, Karadağ ŞG, Çakan M, Argın M, Sezak M. Clinical experiences in Turkish paediatric patients with chronic recurrent multifocal osteomyelitis. Turk J Pediatr 2019; 61: 879-884. Chronic recurrent multifocal osteomyelitis (CRMO) is a clinical entity which occurs mainly in children and adolescents with recurrent episodes of pain occurring over several years. Cause and physiopathology of disease is still uncertain. We aim to assess clinical characteristics and treatment options, need and response to anti-inflammatory therapies in children diagnosed chronic recurrent multifocal osteomyelitis Demographic data and clinical features of seventeen children diagnosed with CRMO in 2 pediatric rheumatology centers in Turkey were reviewed retrospectively. The diagnosis was based on clinical findings, radiological images and histopathological and microbiological studies. A total of 17 patients were included in the study. The median age of diagnosis was 9.6±4.2 years. The mean follow-up time was 31.6 months (range 6-35 months). Most patients (n: 10) had a recurrent multifocal disease course ( > 6 months), 6 patients had a persistent course and a patient had only one episode of CRMO. MEFV gene mutations were detected in 4 patients whose clinical features reduced with colchicine therapy. All patients had received nonsteroidal anti-inflammatory drugs but only one had complete response. Thirteen children with NSAID failure subsequently received corticosteroids, sulfasalazine, methotrexate, Anti TNF α drugs, or a combination of these drugs. This study is the largest cohort of pediatric CRMO patients in our country. Clinical evolution and imaging investigations should be closely done to avoid delays in diagnosis. Ethnic differences create changes in the presentation of the disease and response to treatment.

Keywords: chronic recurrent multifocal osteomyelitis; diagnosis; therapy.

MeSH terms

  • Adolescent
  • Anti-Inflammatory Agents, Non-Steroidal / therapeutic use
  • Child
  • Child, Preschool
  • Female
  • Follow-Up Studies
  • Glucocorticoids / therapeutic use
  • Humans
  • Immunosuppressive Agents / therapeutic use
  • Male
  • Methotrexate / therapeutic use
  • Mutation
  • Osteomyelitis / diagnosis*
  • Osteomyelitis / drug therapy*
  • Pyrin / genetics
  • Retrospective Studies
  • Sulfasalazine / therapeutic use
  • Turkey

Substances

  • Anti-Inflammatory Agents, Non-Steroidal
  • Glucocorticoids
  • Immunosuppressive Agents
  • MEFV protein, human
  • Pyrin
  • Sulfasalazine
  • Methotrexate

Supplementary concepts

  • Chronic recurrent multifocal osteomyelitis