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Chronic recurrent multifocal osteomyelitis: Case report and review of the literature
Lin Liu1, Ranran Zhang, Nana Nie
1Department of Pediatric Nephrology, Rheumatology, and Immunology, The Affiliated Hospital of Qingdao University, Qingdao, China.
Backgrounds:
Chronic recurrent multifocal osteomyelitis (CRMO) is a rare inflammatory disease.
Objective:
This report aims to analyze the clinical characteristics of CRMO and enhance clinicians' comprehension. We present 3 atypical cases, highlighting their unique clinical features, diagnostic challenges, and effective treatment strategies.
Methods:
We retrieved 3 CRMO cases in our hospital from September 2019 to August 2022. The clinical features were analyzed retrospectively, and relevant literatures were reviewed.
Results:
All 3 cases initially presented with bone pain, normal leucocyte counts, negative rheumatoid factors and no signs of sclerotic or hyperostotic lesions. Case 1, a 12-year-old girl, exhibited concurrent acne on the forehead and historic necrotizing lymphadenitis, a previously unreported association with CRMO. Case 2, a 14-year-old boy, tested positive for human leukocyte antigen-B27 and displayed scoliosis along with multifocal osteomyelitis. Case 3, a 9-year-old girl, presented with scoliosis, and chest computed tomography revealed changes in the T8 vertebral body, initially suggesting Langerhans cell histiocytosis. Bone biopsy was conducted in case 1 and case 3, revealing chronic inflammation. All 3 cases affected long bones, pelvis, and vertebra, involving 8, 6 and 5 bones, respectively, identified by magnetic resonance imaging. Genetic analysis was undertaken in cases 1 and 2 but no pathogenic mutations were identified. Upon the confirmation of a CRMO diagnosis, all patients were initiated on a treatment regimen comprising nonsteroidal anti-inflammatory drugs and tumor necrosis factor-α inhibitors. In cases 1 and 2, due to the severity of their bone pain, they were also administered to disease-modifying anti-rheumatic drugs, specifically methotrexate. All 3 patients achieved remission of bone pain. To gain a more comprehensive understanding of CRMO, we conducted a thorough review of relevant literature.
Conclusion:
CRMO is a rare autoinflammatory bone disorder with diverse clinical presentations and a lack of specific laboratory tests, which leads to potency to misdiagnosis or delayed diagnosis. By raising awareness and improving diagnostic criteria, physicians are now better equipped to identify CRMO. We contribute to share our understanding of CRMO by presenting 3 cases with untypical clinical features, highlighting the importance of recognizing this rare condition for timely and effective management.
Insights
Chronic recurrent multifocal osteomyelitis (CRMO) is a rare inflammatory bone disease. This study highlights three atypical CRMO cases, emphasizing unique features and effective management strategies for this challenging condition.
Area of Science:
- Pediatric Rheumatology
- Osteology
- Autoinflammatory Diseases
Background:
- Chronic recurrent multifocal osteomyelitis (CRMO) is a rare, challenging inflammatory bone disorder.
- Its diverse clinical presentations and lack of specific diagnostic markers contribute to diagnostic delays.
- Understanding atypical CRMO manifestations is crucial for timely diagnosis and management.
Purpose of the Study:
- To analyze the clinical characteristics of CRMO.
- To enhance clinicians' comprehension of CRMO through atypical case presentations.
- To highlight diagnostic challenges and effective treatment strategies for CRMO.
Main Methods:
- Retrospective analysis of three CRMO cases diagnosed between September 2019 and August 2022.
- Detailed review of clinical features, diagnostic workup, and treatment outcomes.
- Comprehensive literature review to contextualize findings.
Main Results:
- Three pediatric CRMO cases presented with bone pain, normal inflammatory markers, and negative rheumatoid factors.
- Atypical features included concurrent acne and necrotizing lymphadenitis (Case 1), HLA-B27 positivity and scoliosis (Case 2), and vertebral involvement mimicking Langerhans cell histiocytosis (Case 3).
- All patients achieved remission with nonsteroidal anti-inflammatory drugs, TNF-α inhibitors, and methotrexate (for severe cases).
Conclusions:
- CRMO presents with diverse, often atypical, clinical features, necessitating increased physician awareness.
- Early recognition and improved diagnostic criteria are vital to prevent misdiagnosis and delayed treatment.
- Effective management involves a combination of anti-inflammatory and immunomodulatory therapies, leading to favorable outcomes.
