Chronic Recurrent Multifocal Osteomyelitis Involving the Mandible – A Rare Case Report

2021 ◽  
Vol 45 (4) ◽  
pp. 273-277
Author(s):  
Abrar Alamoudi ◽  
Niranzena Panneer Selvam ◽  
Deeba Kashtwari ◽  
Axel Ruprecht ◽  
Matthew Hansen
Keyword(s):  
Treatment Plan ◽  
Chronic Recurrent Multifocal Osteomyelitis ◽  
Primary Objective ◽  
Bone Lesions ◽  
Radiographic Appearance ◽  
Appropriate Treatment ◽  
Bone Biopsies ◽  
Rare Case Report ◽  
Multifocal Osteomyelitis ◽  
Autoinflammatory Condition

Chronic recurrent multifocal osteomyelitis (CRMO) is an uncommon, aseptic, autoinflammatory condition characterized by multifocal bone lesions with pain, swelling, and frequent exacerbations and remissions. It is noteworthy that these lesions occur without any identifiable etiology or microbiologic finding. The clavicle and metaphyses of the long bones are often involved whereas involvement of the mandible is considered rare. It is usually diagnosed by exclusion of other diseases. As it shares most of its features with the more commonly occurring infective osteomyelitis, patients are often unnecessarily subjected to prolonged courses of antibiotics, serial radiation exposures, and repeated bone biopsies. We present a case of CRMO involving the mandible. Our primary objective is to demonstrate the clinical features of this uncommon disorder, highlighting the radiographic appearance. Familiarity with this condition among radiologists greatly increases the likelihood for early diagnosis and formulating an appropriate treatment plan.

Chronic Recurrent Multifocal Osteomyelitis in Children

1988 ◽  
Vol 29 (5) ◽  
pp. 565-570 ◽  
Author(s):  
W. Mortensson ◽  
G. Edeburn ◽  
M. Fries ◽  
R. Nilsson
Keyword(s):  
Bone Scintigraphy ◽  
Bone Biopsy ◽  
Chronic Recurrent Multifocal Osteomyelitis ◽  
Conventional Radiography ◽  
Radiographic Appearance ◽  
Radiologic Evaluation ◽  
Biopsy Specimens ◽  
Multifocal Osteomyelitis ◽  
Vertebral Bodies ◽  
Bacteria And Fungi

Thirty cases of chronic recurrent multifocal osteomyelitis have been reported in the literature. A radiologic evaluation of thirty-one additional cases, 25 of whom also underwent bone scintigraphy, is presented. Bone biopsy specimens were obtained in 16 patients. Most lesions were located in the metaphyses of the long bones adjacent to the physis and had a characteristic, probably pathognomonic appearance. Extension into the epiphyses was rare. Lesions in the vertebral bodies, clavicle and pelvis had possibly a less specific radiographic appearance. Lesions in short tubular bones were non-specific. Bone scintigraphy had a pratical value in evaluating the global distribution of lesions including asymptomatic lesions and lesions in the spine or pelvis, the latter being somewhat hard to detect with conventional radiography. All biopsies showed acute, subacute or chronic unspecific osteomyelitis, sometimes mixed in the same lesion. Staining for bacteria and fungi was negative.

scholarly journals Searching for Bacterial Pathogens in Pediatric Patients with Chronic Recurrent Multifocal Osteomyelitis Using 16S rRNA Quantitative Real-Time PCR

2017 ◽  
Vol 4 (suppl_1) ◽  
pp. S98-S98
Author(s):  
Justin Searns
Keyword(s):  
16S Rrna ◽  
Real Time ◽  
Real Time Pcr ◽  
Bacterial Pathogens ◽  
Bacterial Load ◽  
Chronic Recurrent Multifocal Osteomyelitis ◽  
Molecular Diagnostic ◽  
Quantitative Real Time Pcr ◽  
Bone Biopsies ◽  
Multifocal Osteomyelitis

Abstract Background Chronic recurrent multifocal osteomyelitis (CRMO) is a rare auto-inflammatory disease in children that causes relapsing episodes of pain. Patients are treated with anti-inflammatory medications or immune-modulating agents. Increasing evidence suggests that CRMO is mediated by dysregulation of the interleukin-1 pathway, not a bacterial source. However, CRMO is often a diagnosis of exclusion, and patients occasionally receive antimicrobials for possible culture negative infectious osteomyelitis. Few prior studies have utilized molecular diagnostic techniques to identify bacterial pathogens in CRMO bone biopsies. Methods Musculoskeletal specimens sent for culture during routine clinical care were banked from patients admitted to Children’s Hospital Colorado from 6/2012 to 10/2016. On retrospective chart review, 28 specimens were collected from 16 patients ultimately diagnosed with CRMO. Specimens were processed and extracted prior to molecular testing. All samples underwent quantitative real-time PCR (qPCR) testing using bacterial load assays targeting the bacterial 16S rRNA gene. Results Mean age at time of sample collection was 9.2 years. CRMO diagnosis was made by clinical, pathologic, and radiographic findings. All patients had pathology findings consistent with CRMO including lymphoplasmacytic infiltrate, focal necrosis, and/or marrow fibrosis. All patients had MRI findings consistent with CRMO. No patient had bacteria identified on Gram stain; 2/28 samples (7%) had bacterial growth on culture (both were coagulase-negative staphylococcus, felt to be contaminant). None of the 28 specimens met the threshold of bacterial load on qPCR testing to necessitate bacterial sequencing. None of the 16 patients were treated with antimicrobials and there were no readmissions for clinical worsening. Conclusion CRMO patients did not have bacteria identified on universal bacterial 16S rRNA testing. This finding further supports that CRMO patients do not require antimicrobial therapy. Future steps to exclude infectious pathogens in CRMO could include next-generation DNA sequencing. Disclosures All authors: No reported disclosures.

scholarly journals Zoledronate Associated Seizure in Chronic Recurrent Multifocal Osteomyelitis

2021 ◽  
Author(s):  
Saoussen Miladi ◽  
Yasmine Makhlouf ◽  
Alia Fazaa ◽  
Mariem Sallemi ◽  
Selma Chekili ◽  
...  
Keyword(s):  
Inflammatory Disease ◽  
Chronic Recurrent Multifocal Osteomyelitis ◽  
Clonic Seizure ◽  
Bone Lesions ◽  
Multifocal Osteomyelitis ◽  
Auto Inflammatory Disease

Chronic recurrent multifocal osteomyelitis (CRMO) is an auto-inflammatory disease characterized by sterile bone lesions. We report a case of a patient with CRMO who developed a seizure post bisphosphonate administration. Although, the treatment of CRMO is currently not codified, the most promising results have been observed in patients under treatment with bisphosphonates. Keywords: CRMO; Bisphosphonate; tonico-clonic seizure.

SAPHO and CRMO: The Value of Imaging

2018 ◽  
Vol 22 (02) ◽  
pp. 207-224 ◽  
Author(s):  
Rikke Klicman ◽  
Paolo Simoni ◽  
Philip Robinson ◽  
James Teh ◽  
Anne Jurik
Keyword(s):  
Interdisciplinary Approach ◽  
Chronic Recurrent Multifocal Osteomyelitis ◽  
Imaging Features ◽  
Invasive Procedures ◽  
Palmoplantar Pustulosis ◽  
Appropriate Treatment ◽  
Pathologic Features ◽  
Skeletal Site ◽  
Characteristic Imaging ◽  
Multifocal Osteomyelitis

AbstractThe syndromes synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) and chronic recurrent multifocal osteomyelitis (CRMO) constitute a group of chronic relapsing inflammatory osteoarticular disorders with frequently associated skin eruptions such as palmoplantar pustulosis and acne conglobata and rather characteristic imaging features in the form of osteitis and/or hyperostosis. CRMO predominantly occurs in children/adolescents and SAPHO in adults. Any skeletal site can be involved, and the imaging appearances vary, depending on the patient's age and the stage/age of the lesion. The diagnosis may be difficult if there is no skin disease, but attention to characteristic imaging appearances may help avoid misdiagnosis (e.g., infection and tumor) and thereby unnecessary invasive procedures as well as facilitating early diagnosis and appropriate treatment. This article provides an overview of the radiologic appearances of SAPHO/CRMO and relevant pathogenetic, clinical, and pathologic features to facilitate the diagnosis that often requires an interdisciplinary approach including radiologists.

Chronic recurrent multifocal osteomyelitis: A rare disorder presenting as multifocal bone lesions

2001 ◽  
Vol 37 (2) ◽  
pp. 132-137 ◽  
Author(s):  
Rob Tingley ◽  
Taj Jadavji ◽  
Graham Boag ◽  
Gerhard N. Kiefer ◽  
Cynthia Trevenen ◽  
...  
Keyword(s):  
Chronic Recurrent Multifocal Osteomyelitis ◽  
Rare Disorder ◽  
Bone Lesions ◽  
Multifocal Osteomyelitis

scholarly journals A potential new target for autoinflammatory bone disease

2020 ◽  
Vol 295 (11) ◽  
pp. 3401-3402
Author(s):  
Elizabeth L. Hartland
Keyword(s):  
Mouse Model ◽  
Tyrosine Kinase ◽  
Bone Disease ◽  
Therapeutic Target ◽  
Inflammatory Cytokine ◽  
Chronic Recurrent Multifocal Osteomyelitis ◽  
Bone Lesions ◽  
Genetic Mouse Model ◽  
Multifocal Osteomyelitis ◽  
Key Driver

Chronic recurrent multifocal osteomyelitis (CRMO) is an autoinflammatory bone disease mediated by the inflammatory cytokine, IL-1β. Although IL-1β is known as the key driver of bone lesions in CRMO, the signaling events leading to pathogenic levels of the cytokine are not fully understood. Using a genetic mouse model of CRMO, Dasari et al. find a role for the nonreceptor spleen tyrosine kinase (SYK) in upstream signaling leading to IL-1β up-regulation. Their findings suggest that SYK may constitute a new therapeutic target for CRMO.

scholarly journals Early diagnosis and response assessment in chronic recurrent multifocal osteomyelitis: changes in lesion volume and signal intensity assessed by whole-body MRI

2021 ◽  
Author(s):  
Angelina Kieninger ◽  
Jürgen F. Schäfer ◽  
Ilias Tsiflikas ◽  
Monika Moll ◽  
Jasmin Kümmerle-Deschner ◽  
...  
Keyword(s):  
Early Diagnosis ◽  
Chronic Recurrent Multifocal Osteomyelitis ◽  
Response Assessment ◽  
Quantitative Mri ◽  
Whole Body ◽  
Bone Lesions ◽  
Lesion Volume ◽  
Mri Features ◽  
Multifocal Osteomyelitis

Objective: To assess the effectiveness of whole-body MRI (WB-MRI) in early diagnosis of chronic recurrent multifocal osteomyelitis (CRMO) and the prediction of clinical response through quantitative MRI features. Methods: 20 children (mean age, 10.3 years; range, 5–14 years) with CRMO underwent WB-MRI and were assessed with a clinical score (Jansson) at baseline (median time after first encounter, 8 months) and follow-up (median time after baseline, 11.5 months). Baseline WB-MRI scans were classified as early (within 6 months after first encounter) and late. Clinical responders and non-responders were compared regarding number and localization of bone lesions, lesion volume and T2 signal intensity (SI) ratio (lesion to muscle). Results: Diagnosis of CRMO was made promptly in the early WB-MRI group (n = 10; median, 3 months) compared to the late WB-MRI group (n = 10; 18 months; p = 0.006). Bone lesions were mainly located in the lower extremities (n = 119/223; 53%). No significant difference was detected regarding the number of bone lesions and lesion volume in the subgroups of clinical responders (n = 10) and non-responders (n = 10). Responders showed a higher volume reduction of bone lesions at follow-up compared to non-responders (p = 0.03). Baseline and follow-up SI ratios were lower in responders (5.6 and 5.8 vs 6.1 and 7.2; p = 0.047 and p = 0.005). Conclusion: The use of WB-MRI within 6 months of disease suspicion may serve as a benchmark to support early diagnosis of CRMO. T2 SI ratios and the reduction of lesions’ volume correlate with clinical outcome. Advances in knowledge: WB-MRI at an early stage of suspected CRMO plays a key role for early diagnosis. This is the first study showing that quantitative MRI features are suitable for response assessment and can be used as prognostic markers for the prediction of clinical response.

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