Background/Purpose: Chronic Recurrent Multifocal Osteomyelitis (CRMO) is an autoinflammatory bone disease requiring immunosuppressive therapy in half of patients. Monoclonal Tumor Necrosis Factor inhibitors (TNFi) are second-line off-label therapies, with undesired paradoxical psoriasisoccurring in a subset of patients on TNFi. This can prompt conversion to alternate therapy which can prove challenging, given few TNFi are approved for use in children.

Objective:To determine the efficacy and safety of golimumab, a fully humanized TNFi, in children with CRMO, including those with paradoxical psoriasis following exposure to other monoclonal TNFi.

Methods: A single center retrospective chart review of patients with CRMO who received golimumab between June 1, 2018, and December 21, 2020, was conducted. Patients diagnosed < 21 years old with ³ 1 follow-up and ³ 3 months of treatment with golimumab were included.

Data including whole-body MRI (WB-MRI) lesion counts, prior treatment, concomitant medications, demographics, clinically relevant data, laboratory results, patient-reported outcomes (PRO), pain scores, and psoriasis burden were extracted. Linear mixed models with log-transformed outcomes were used to assess changes over time with a random effect included to account for within-subject correlation of repeated measures. Confidence intervals of 95% and p-values were reported and considered significant if ≤ 0.05.

Results: Eighteen patients were included, fourteen of whom were previously treated with disease-modifying antirheumatic drugs (DMARDs) and seventeen receiving other TNFi. Medianbone lesion count, physician global assessments, pain scores, and erythrocyte sedimentation rate decreasedat 3- and 6-months follow-up. Five of the nine patients with baseline paradoxical psoriasis had improvement or resolution of paradoxical psoriasis over time. Two patients had worsening of baseline paradoxical psoriasis and one patient who did not previously have TNFi-induced psoriasis developed psoriasis at 3-months. One patient discontinued therapy at 4 months given lack of radiographic improvement and persistence or worsening of pre-existing paradoxical psoriasis. Two children received dual therapy with both golimumab and ustekinumab with clinical improvement. No serious infections or adverse events (AE) were observed during this study.

Conclusion: Golimumab is safe and effective to treat children with CRMO with a low risk of paradoxical psoriasis and improvement in baseline paradoxical psoriasis induced by other TNFi. Additional long-term surveillance with larger sample size is needed to further characterize the safety and efficacy of golimumab and overall risk of paradoxical psoriasis.

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ACR Meeting Abstracts - https://acrabstracts.org/abstract/golimumab-therapy-in-children-with-chronic-recurrent-multifocal-osteomyelitis-a-case-series-reviewing-safety-and-efficacy/