Canakinumab treatment in a young girl with refractory chronic recurrent multifocal osteomyelitis associated with pyoderma gangrenosum

Int J Rheum Dis. 2022 Nov;25(11):1333-1338. doi: 10.1111/1756-185X.14425. Epub 2022 Aug 24.


Background: Chronic recurrent multifocal osteomyelitis (CRMO) is a bone inflammatory disorder characterized by osteolytic, usually multiple, symmetric lesions. Diagnosis is one of exclusion, and no standardized therapies are available. Presumed deregulation of the interleukin (IL)-1β axis, as observed in 2 monogenic autoinflammatory conditions such as Majeed syndrome (LPIN2 mutations) and deficiency of IL-1 receptor antagonist (IL1RN mutations) with CRMO-like bone involvement, suggests the blockade of IL-1 as potentially useful also in this condition, even if scarce data are available.

Case presentation: We report the case of a 13-year-old girl affected by a multidrug-resistant and pyoderma gangrenosum-complicated CRMO treated with canakinumab, a human monoclonal antibody targeting IL-1β.

Conclusion: In this young patient pyoderma gangrenosum and CRMO showed a rapid and satisfactory response to canakinumab, although over time a decreased efficacy in controlling bone disease was observed.

Keywords: IL-1; anakinra; canakinumab; chronic recurrent multifocal osteomyelitis; pyoderma gangrenosum.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Anemia, Dyserythropoietic, Congenital* / complications
  • Anemia, Dyserythropoietic, Congenital* / genetics
  • Female
  • Humans
  • Osteomyelitis* / complications
  • Pyoderma Gangrenosum*


  • canakinumab

Supplementary concepts

  • Chronic recurrent multifocal osteomyelitis