Biologic treatment for chronic recurrent multifocal osteomyelitis: report of four cases and review of the literature

Tronconi, Elena; Miniaci, Angela; Baldazzi, Michelangelo; Greco, Laura; Pession, Andrea

doi:10.1007/s00296-017-3877-0

Cited by 19 publications

(9 citation statements)

References 28 publications

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“…[20] Longitudinal assessment of changes in radiological disease activity in children with CNO has previously predominantly been descriptive, mainly reporting the number of bone lesions and changes in bone marrow oedema based on expert opinion. [3,8,46,47,29,31,32,36,[42][43][44][45]. Changes in radiological disease activity following pamidronate therapy have been reported based on a WBMRI scoring system encompassing the number, relative size and signal intensity of active CNO lesions as well as soft tissue in ammation and structural lesions in the form of osseous hyperostosis and vertebral collapse, but the study was primarily performed to test reliability of the MR assessment and did not include global patient scores for disease severity.…”

Section: Discussionmentioning

confidence: 99%

Standardized Reporting and Quantification of Whole-Body MRI findings in Children with Chronic Non-Bacterial Osteomyelitis Treated with Pamidronate

Andreasen

Klicman

Herlin

et al. 2022

Preprint

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ObjectivesTo test a modified radiological index for non-bacterial osteitis (mRINBO) used for standardized reporting and quantification of whole-body MRI (WBMRI) findings in children with chronic non-bacterial osteomyelitis (CNO) receiving pamidronate therapy.MethodsWBMRI was assessed in 18 children with CNO at baseline and after receiving pamidronate therapy for one year. Parameters of interest were: number and anatomic site of radiologically active bone lesions (RAL), size of RAL, extramedullary affection, spinal involvement and changes in mRINBO, which includes both the number and maximal size of RAL (RALmax) in addition to extramedullary and chronic changes. ResultsAt the time of diagnosis, the mean age of the children was 9.8 (sd, 8.7-10.9) years and 11/18 were females. The number of RALs per patient decreased from median [interquartile range] 4.5 [3-8] to 3 [2-5] RALs per patient (p=0.02) and extramedullary inflammatory changes regressed. Sixty-one percent of all RALs occurring at baseline resolved and three children became without active inflammatory lesions by WBMRI. However, the median size of RALs did not change when taking new lesions occurring in 7/18 children into account, but RALmax decreased significantly from 39 [29-45] mm at baseline to 28 [20-40] mm (p<0.01) at year-one with a concomitant decrease of mRINBO from a median of 5 [4-7] to 4 [3-5] (p=0.05).ConclusionsmRINBO decreased during pamidronate therapy and mRINBO may be a potential scoring method to quantify changes in radiological disease activity in children with CNO. Further studies are needed to test feasibility and validity of mRINBO.

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Section: Discussionmentioning

confidence: 99%

Standardized Reporting and Quantification of Whole-Body MRI findings in Children with Chronic Non-Bacterial Osteomyelitis Treated with Pamidronate

Andreasen

Klicman

Herlin

et al. 2022

Preprint

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“…Treatment guidelines for CRMO are still under discussion and include watchful waiting for spontaneous remission, although the therapy of first choice consist of nonsteroidal anti-inflammatory drugs [28]. Glucocorticosteroids, bisphosphonates including pamidronate, sulfasalazine, anti-tumor necrosis factor α (TNFα) drugs have been used with good results (Table 1) [25–27]. Antibiotic treatment is considered ineffective [2, 6].…”

Section: Discussionmentioning

confidence: 99%

A complicated path to the CRMO diagnosis – case of a 9 year old girl whose story comes full circle

Świdrowska-Jaros

Smolewska

2019

BMC Musculoskelet Disord

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Background Chronic recurrent multifocal osteomyelitis (CRMO) is a rare idiopathic autoinflammatory bone disease that mostly affects children and adolescents. It is a diagnosis of exclusions since no clinical signs and symptoms are pathognomonic. Radiological tests are often essential, but bone biopsy may be needed in unclear cases. Case presentation A 9-year-old Caucasian girl with a history of bone pain. The data from the history and results of laboratory tests suggested osteomyelitis, but no adequate response to the treatment was observed. A number of imaging tests did not confirm the diagnosis, therefore a bone biopsy was necessary. Conclusions Differential diagnosis of CRMO is challenging and it is based on exclusions. Since it might be misdiagnosed or mistreated, bone biopsy should be considered in patients reporting bone pain who are unresponsive to treatment.

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“…53 Biological agents such as etanercept and infliximab act by their inhibition of tissue necrosis factor and have been demonstrated to improve clinical symptoms but lack large, long-term data. 54…”

Section: Discussion and Literature Reviewmentioning

confidence: 99%

Chronic Recurrent Multifocal Osteomyelitis of the Hand: A Rare Pediatric Condition

Karunaratne

Davies

Carty

et al. 2019

Hand (New York, N,Y.)

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Background: Chronic recurrent multifocal osteomyelitis (CRMO) is a rare autoinflammatory condition characterized by bone pain and swelling, secondary to sterile bone osteolytic lesions. Estimated incidence is 1:1 000 000, with ~2% involving the hand. We present a case series of CRMO of the hand and review the available literature, with the aim of educating and raising awareness of this condition. Methods: A retrospective chart review was conducted for all patients presenting to the institution diagnosed with CRMO involving the hands. Subsequently, a literature review was performed. Results/Case Series: Three cases of CRMO of the hand were identified. Age ranged from 20 months to 6 years, with 1 female. Time from presentation to diagnosis ranged from 3 to 15 months. Two had a single lesion, with the remaining patient having a second lesion on full-body magnetic resonance imaging (MRI). Three lesions involved phalanges, and one involved a metacarpal. Two received antibiotics without improvement prior to diagnosis. After diagnosis of CRMO, two received intravenous pamidronate. They both later showed improvement on MRI. Conclusion: CRMO is a rare disease with scarce literature to guide management. We present a series of CRMO specifically of the hand. The cases educate key stakeholders, raise awareness of the diagnosis, and illustrate challenging aspects of managing these patients. This includes the special functional and anatomical consideration essential in managing cases involving the hand.

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Biologic treatment for chronic recurrent multifocal osteomyelitis: report of four cases and review of the literature

Cited by 19 publications

References 28 publications

Standardized Reporting and Quantification of Whole-Body MRI findings in Children with Chronic Non-Bacterial Osteomyelitis Treated with Pamidronate

Standardized Reporting and Quantification of Whole-Body MRI findings in Children with Chronic Non-Bacterial Osteomyelitis Treated with Pamidronate

A complicated path to the CRMO diagnosis – case of a 9 year old girl whose story comes full circle

Chronic Recurrent Multifocal Osteomyelitis of the Hand: A Rare Pediatric Condition

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