How Have the Diagnosis and Treatment of Chronic Recurrent Multifocal Osteomyelitis Changed Over Time?

Abstract

Purpose: Chronic recurrent multifocal osteomyelitis (CRMO) is a rare inflammatory condition characterized by sterile bone lesions. There appears to be a shift in the diagnostic modalities and treatment over the past decades despite insufficient published data. The purpose of this study was to document: 1) the number of patients diagnosed with CRMO, 2) patient demographics, 3) disease characteristics at presentation, 4) diagnostic modalities employed, and 5) treatments prescribed at our institution over a 30-year period.

Methods: This single-center, retrospective cohort study included children diagnosed with CRMO who presented between 1990 and 2020. The electronic medical records were queried using numerous search terms. Patients were excluded if CRMO was included in the differential diagnosis but was not confirmed at the time of chart review or if CRMO was suspected early in the disease course but the patient was ultimately diagnosed with another condition. The predictor (time in years) and outcome variables (diagnostic modalities and treatment types) were tested using bivariate analyses using IBM SPSS, Version 27 (IBM Corp., Armonk, NY).

Results: A total of 224 patients were diagnosed with CRMO during the observation period (68.3% female; 67.4% white). The number of patients diagnosed over the past decade rose by 215%, as compared to the previous 2 decades (1990 to 2010). Regional magnetic resonance imaging (83.8%) and biopsy (66.5%) were the most utilized diagnostic modalities over the past decade, with a statistically significant decline in the proportion of biopsies performed (66.5% during the past decade vs 84.9% in the previous 2 decades, P = .01). Over the past decade, nonsteroidal anti-inflammatory drugs (40.1%), disease-modifying antirheumatic drugs (27.1%), and tumor necrosis factor inhibitors (21.1%) were the most commonly used treatments, with a statistically disproportionate increase in the use of tumor necrosis factor inhibitors (21.1% during the past decade vs 3.8% in the previous 2 decades, P < .001).

Conclusions: This is one of the largest CRMO cohort studies and the only study to observe changes in diagnostic modalities and treatment over a 30-year period. Future studies should assess the impact of variations in clinical presentation, time to diagnosis, diagnostic modalities, and management as predictors of disease outcomes.