Exploring the Role of Canakinumab in the Treatment of Autoinflammatory Bone Disorders: A Systematic Review.
Background: Autoinflammatory bone disorders are rare, non-infectious inflammatory conditions that primarily involve the skeleton, most commonly presenting as chronic nonbacterial osteomyelitis (CNO) or chronic recurrent multifocal osteomyelitis (CRMO). Less frequently, they occur in the context of M...
| Published in: | Rheumato Vol. 6; no. 1; pp. 6 - 15 |
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| Main Authors: | , |
| Format: | research systematic review tables/charts Journal Article |
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MDPI
Mar2026
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| Online Access: | View this record in EBSCOhost |
| fields | @attributes: recordID: 1 pdfLink: plink: https://search.ebscohost.com/login.aspx?direct=true&db=ccm&AN=192662484&site=ehost-live header: @attributes: shortDbName: ccm uiTerm: 192662484 longDbName: CINAHL Complete uiTag: AN controlInfo: bkinfo: dissinfo: jinfo: jid: 26740621 N1MW jtl: Rheumato issn: 26740621 maglogo: N pubinfo: dt: Mar2026 vid: 6 iid: 1 pid: 97109 pub: MDPI artinfo: ui: 192662484 192662484 192662484 10.3390/rheumato6010006 192662484 ppf: 6 ppct: 9 formats: tig: atl: Exploring the Role of Canakinumab in the Treatment of Autoinflammatory Bone Disorders: A Systematic Review. aug: au: Gamalero, Lisa Giani, Teresa affil: Pediatric Unit, ULSS2 Marca Trevigiana, Conegliano Hospital, 31015 Conegliano, Italy sug: subj: Osteomyelitis Drug Therapy Hereditary Autoinflammatory Diseases Drug Therapy Antibodies, Monoclonal Therapeutic Use Antibodies, Monoclonal Metabolism Drug Efficacy Evaluation Patient Safety Human Systematic Review PubMed Medline Cochrane Library Interleukins Blood Antibodies, Monoclonal Administration and Dosage Phenotype Osteoclasts ab: Background: Autoinflammatory bone disorders are rare, non-infectious inflammatory conditions that primarily involve the skeleton, most commonly presenting as chronic nonbacterial osteomyelitis (CNO) or chronic recurrent multifocal osteomyelitis (CRMO). Less frequently, they occur in the context of Mendelian syndromes such as Majeed syndrome, deficiency of the interleukin-1 receptor antagonist (DIRA), and pyogenic arthritis; pyoderma gangrenosum; and acne (PAPA) syndrome. Given the role of IL-1-driven innate immune dysregulation across these bone disorders, and the growing, though heterogeneous, clinical experience with IL-1 blockade, this review maps and critically appraises the available evidence on canakinumab in autoinflammatory bone disorders. Methods: We systematically searched PubMed and the Cochrane Library (English, inception–July 2025) and screened ClinicalTrials.gov. Eligible reports included any case reports/series describing canakinumab use in autoinflammatory bone disorders (CNO/CRMO, Majeed, DIRA, PAPA). Results: Six publications met the inclusion criteria (one case series, five case reports; 10 patients). Complete responses were reported in all three patients with Majeed syndrome and in two patients with sporadic CRMO associated with systemic features. Partial responses occurred in two additional sporadic CRMO cases, while no meaningful response was documented in DIRA. No interventional trials of canakinumab were identified on ClinicalTrials.gov for CNO/CRMO, Majeed, DIRA, or PAPA. Conclusions: Although the role of IL-1 in the pathogenesis of autoinflammatory bone disease provides a rationale for IL-1 blockade, evidence for canakinumab remains limited and heterogeneous, precluding definitive conclusions. Indicators of benefits appear most consistently in Majeed syndrome and in selected CRMO phenotypes. pubtype: Academic Journal doctype: research systematic review tables/charts Journal Article ougenre: Article language: English refInfo: holdings: @attributes: islocal: N |
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