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Canakinumab treatment in a young girl with refractory chronic recurrent multifocal osteomyelitis associated with
Sabrina Acierno1, Francesca Angrisani1, Achille Marino2
1Department of Woman, Child and of General and Specialized Surgery, Università degli Studi della Campania "Luigi Vanvitelli", Naples, Italy.
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.
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