Canakinumab treatment in a young girl with refractory chronic recurrent multifocal osteomyelitis associated with pyoderma gangrenosum.
Int J Rheum Dis
; 25(11): 1333-1338, 2022 Nov.
Article
de En
| MEDLINE
| ID: mdl-36004431
ABSTRACT
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)-1ß 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-1ß.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.Mots clés
Texte intégral:
1
Collection:
01-internacional
Base de données:
MEDLINE
Sujet principal:
Ostéomyélite
/
Pyodermie phadégénique
/
Anémie dysérythropoïétique congénitale
Type d'étude:
Risk_factors_studies
Limites:
Adolescent
/
Female
/
Humans
Langue:
En
Journal:
Int J Rheum Dis
Sujet du journal:
REUMATOLOGIA
Année:
2022
Type de document:
Article
Pays d'affiliation:
Italie