Clinical features and diagnostic approaches to chronic recurrent multifocal osteomyelitis: A retrospective study

Abstract Background: Chronic recurrent multifocal osteomyelitis (CRMO) is an autoinflammatory bone disease which is difficult to diagnose in the early stages. The aim of this study was to summarize the methods of diagnosis and differential diagnosis of CRMO, study diagnostic methods for early identification and diagnosis of the disease, and improve the ability of clinicians to identify and diagnose the disease at an early stage. Methods: All children diagnosed with CRMO at our hospital between 2013 and 2022 were included. Clinical characteristics, imaging findings, and reasons for delayed diagnoses were summarized. In addition, the literature on CRMO from database establishment to February 1, 2023, was retrieved through Embase, Medline, and PubMed, and the management of CRMO was proposed based on the literature. Results: There were seven patients with CRMO (six males and one female). Mean age at first symptom: 6.3 (IQR 2.3-8.2) years; mean delay in diagnosis (from symptoms): 2.1 (IQR 1.2-2.8) years; mean age at relapse: 8.1 (IQR 3.2-12.1) years; mean disease duration: 3.8 (IQR 2.4-3.9) years. All children had local swelling and bone pain, and three patients had pathological fractures (one in the femur and two in the tibia). A total of 34 anatomical sites were involved in the lesions in seven children; one case had a single lesion, and six had multiple lesions. The blood and puncture tissue bacterial culture results in seven cases were negative, and the bone marrow cytology examination was normal. A total of 119 clinical research reports on CRMO (1202 patients) were included, and it was found that the first symptom in 1142 cases (approximately 95%) was bone pain or local swelling. In all cases in which bacterial cultures of peripheral blood or bone tissue were performed, the results were negative. In addition, bone marrow cytology results were negative in 1178 (98%). Conclusions: This study systematically summarized the clinical characteristics of patients with CRMO in our center and those reported in the literature. In addition, this study proposes primary and secondary diagnostic criteria for the management of CRMO, which requires verification.