The Many Faces of Pediatric Chronic Recurrent Multifocal Osteomyelitis (<scp>CRMO)</scp>: A Practical Location‐ and Case‐Based Approach to Differentiate <scp>CRMO</scp> From Its Mimics

Menashe, Sarah J.; Aboughalia, Hassan; Zhao, Yichao; Ngo, Anh-Vu; Otjen, Jeffrey P.; Thapa, Mahesh; Iyer, Ramesh S.

doi:10.1002/jmri.27299

Cited by 22 publications

(13 citation statements)

References 67 publications

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“…A minority of patients with CNO are found to have IBD. At the time of writing, there have been only 25 case reports of IBD-associated CNO among children and this is the first report in a child with ASD, for whom developmental/language delay and suspected nutritional deficiencies complicated the diagnosis ( 18 – 24 ). Among patients with IBD-associated CNO, it is common for CNO symptoms to precede gastrointestinal symptoms, which may also contribute to diagnostic delay ( 18 ).…”

Section: Discussionmentioning

confidence: 97%

Chronic Non-infectious Osteomyelitis Mimicking Scurvy as the Presenting Sign of Crohn’s Disease: Case Report

et al. 2022

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Chronic non-infectious osteomyelitis (CNO) is a rare, inflammatory process associated with pediatric inflammatory bowel disease (IBD). Signs and symptoms of CNO parallel scurvy, a nutritional deficiency that can affect children with autism spectrum disorder (ASD). This is the first report of a child initially thought to have scurvy, then subsequently diagnosed with CNO as the presenting manifestation of Crohn’s disease. This case enhances the literature elucidating extra-intestinal manifestations of IBD and pediatric nutritional deficiencies.

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Section: Discussionmentioning

confidence: 97%

Chronic Non-infectious Osteomyelitis Mimicking Scurvy as the Presenting Sign of Crohn’s Disease: Case Report

et al. 2022

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“…However, all of our patients had no less than 2 bone lesions, which might be related to the long disease course. It is worth noting that CNO is an exclusionary diagnosis and is important to distinguish from infections and tumors ( 2 ). We suggest that bone biopsy and thorough etiological examination (smear and culture) are very necessary for the differential diagnosis ( 22 ).…”

Section: Discussionmentioning

confidence: 99%

Clinical and Genetic Features of Chinese Adult Patients With Chronic Non-Bacterial Osteomyelitis: A Single Center Report

Zhao

et al. 2022

Front. Immunol.

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ObjectivesChronic non-bacterial osteomyelitis (CNO) is a rare polygenic autoinflammatory bone disease. We aimed to characterize the clinical manifestations and gene variants of Chinese adult patients with CNO.MethodsBy reviewing data of all CNO patients being diagnosed and followed up at the Center for Adult Autoinflammation Diseases, Department of Rheumatology, Peking Union Medical College Hospital, clinical and genetic features of these patients were evaluated and concluded.ResultsThe median age of disease onset was 19 (6-64) years old, and adult-onset was observed in 6 (60%) patients. The mean time of diagnosis delay was 92 ± 78 months. The common symptoms were bone pain (10, 100%), fever (9, 90%), and arthritis (6, 60%). In total, there were 54 skeletal lesions, and each patient had no less than 2 lesions. The most frequently affected sites included lower limbs (20.5%), mandible, vertebrae and pelvis (17.5%, separately). Variants of 4 genes were detected in our study including COL1A1, PSTPIP1, LRP5 and CLCN7. In seven patients who were treated with combination therapy containing tumor necrosis factor (TNF) α inhibitors, five (55.6%) had a complete response and 2 (44.4%) had a partial response.ConclusionThis is the first and largest case series of CNO in the Chinese adult patients. Four novel genetic mutations potentially associated with CNO were identified. Notably, CNO should be considered in the differential diagnosis of adult patients with long disease course and recurrent multifocal osteomyelitis of unknown cause, and these patients might benefit from combination therapy containing TNFα inhibitors.

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“…Having described the typical from of CRMO involvement, the disease can manifest a variety of presentations (9). There have been many case reports and case series of patients with atypical involvement (4,10,11).…”

Section: Discussionmentioning

confidence: 99%

“…There have been many case reports and case series of patients with atypical involvement (4,10,11). For instance, patients with acute vertebral deformities (12), chronic painful scoliosis (13), and chronic mandible involvement as their only presentation (9). This variety has been considerable, to the point that some studies have proposed classi cation of CRMO cases into subgroups based on their presented phenotypes, such as spinal or axial versus peripheral form (14,15).…”

Section: Discussionmentioning

confidence: 99%

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Chronic Recurrent Multifocal Osteomyelitis with an uncommon presentation and disabling features: A case report

Shahin

Asnaashari

Zarei

et al. 2022

Preprint

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Background: Chronic recurrent multifocal osteomyelitis (CRMO) is one of the auto-inflammatory bone disorders with yet to be determined etiology. Due to overlapping signs and symptoms with other bone involvements, the diagnosis of this disease might take a long period of time and the patients may develop deformities in long-term. Thus, increasing awareness about different presentations of the disease seems beneficial.Case presentation: A 12-year-old boy was referred to our center with severe back pain in his thoracic vertebrae from 18 months before referral. He had previously undergone two vertebroplasty surgeries due to his kyphosis and had received several courses of antibiotics, which resulted in no improvement in his disabling pain. His physical examination was unremarkable, except for severe thoracic vertebrae tenderness and a mild dextroscoliosis. MRI showed diffuse multiple hypo-signal lesions in thoracic vertebral bodies with slight enhancement, causing compression fractures of T8, T11 and T12 bodies. Bone scan revealed increased activity in thoracic region (T7-T8-T9-T12 vertebral bodies) and left clavicle. Other differentials were ruled out by bone marrow aspiration, bone biopsy and laboratory tests. His disease showed a favorable response to treatment with Pamidronate, Methotrexate and Indomethacin and he had no relapse in three years of follow-up.Conclusions: Uncommon presentations of CRMO can be misleading to the specialists and might result in prolonged diagnosis process. Increased awareness among different specialists about such presentations can potentially lead to earlier diagnosis, and timelier and more effective treatment of the CRMO patients.

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The Many Faces of Pediatric Chronic Recurrent Multifocal Osteomyelitis (CRMO): A Practical Location‐ and Case‐Based Approach to Differentiate CRMO From Its Mimics

Cited by 22 publications

References 67 publications

Chronic Non-infectious Osteomyelitis Mimicking Scurvy as the Presenting Sign of Crohn’s Disease: Case Report

Chronic Non-infectious Osteomyelitis Mimicking Scurvy as the Presenting Sign of Crohn’s Disease: Case Report

Clinical and Genetic Features of Chinese Adult Patients With Chronic Non-Bacterial Osteomyelitis: A Single Center Report

Chronic Recurrent Multifocal Osteomyelitis with an uncommon presentation and disabling features: A case report

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