Consensus Treatment Plans for Chronic Nonbacterial Osteomyelitis Refractory to Nonsteroidal Antiinflammatory Drugs and/or With Active Spinal Lesions

Zhao, Yong; Wu, Eveline Y.; Oliver, Melissa; Cooper, Ashley M.; Basiaga, Matthew L.; Vora, Sheetal S.; Lee, Tzielan; Fox, Emily; Amarilyo, Gil; Stern, Sara; Dvergsten, Jeffrey; Haines, Kathleen A.; Rouster‐Stevens, Kelly; Onel, Karen; Cherian, Julie; Hausmann, J.; Miettunen, Päivi; Cellucci, Tania; Nuruzzaman, Farzana; Taneja, A.; Barron, Karyl S.; Hollander, Matthew; Lapidus, Sivia; Li, Suzanne C.; Özen, Seza; Girschick, Hermann; Laxer, Ronald M.; Dedeoğlu, Fatma; Hedrich, Christian M.; Ferguson, Polly J.; Osteomyelitis, Chronic Nonbacterial

doi:10.1002/acr.23462

Cited by 138 publications

(132 citation statements)

References 37 publications

Supporting

Mentioning

126

Contrasting

Unclassified

Order By: Relevance

“…The Childhood Arthritis and Rheumatology Research Alliance (CARRA) have developed three Consensus Treatment Plans for the treatment of CNO in patients refractory to NSAIDs and/or with active spinal lesions with either methotrexate or sulfasalazine, TNF blockers (with or without methotrexate) or bisphosphonates. Use of these Consensus Treatment Plans will provide more information on efficacy in the absence of randomised control trials [ 14 ].…”

Section: Discussionmentioning

confidence: 99%

Chronic non bacterial osteitis- a multicentre study

et al. 2018

View full text Add to dashboard Cite

ObjectiveTo understand the demographics, clinical features and treatment outcomes of Chronic Non-bacterial Osteitis (CNO) from three tertiary paediatric rheumatology services in the United Kingdom.MethodsChildren less than 18 years of age diagnosed with CNO between 2001 to 2016 from one tertiary service and between 2001 to 2017 from two tertiary services were included. Clinical notes were reviewed and all pertinent data were collected on a pre-defined proforma. One hundred and thirty one patients were included in the study. The Bristol diagnostic criteria were applied retrospectively.ResultsRetrospective analysis of the data showed that the disease was more common in girls than boys (2.5:1), median age at onset of symptoms was 9.5 years (IQR 8 to 11 years). Bone pain was the predominant symptom in 118/129 (91.4%) followed by swelling in 50/102 (49.01%). Raised inflammatory markers were present in 39.68% of the patients. Whole body Magnetic Resonance Imaging (MRI) was a useful diagnostic tool. Metaphyses of long bones were most often involved and the distal tibial metaphyses 65/131 (49.6%) was the most common site. Non-steroidal anti-inflammatory drugs were used as first line (81.67%) followed by bisphosphonates (61.79%). Treatment was escalated to a TNF blocker when response to bisphosphonates was suboptimal. The disease was in remission in 82.4% of the patients during the last follow up.ConclusionOur multicentre study describes features and outcomes of CNO in a large number of patients in the United Kingdom.Significance and innovation Raised inflammatory markers were present in 39.68% of our patients.Whole body MRI is useful for diagnosis and also determining response to treatment.A greater number of lesions were detected on radiological imaging compared to clinical assessment.Metaphyses of long bones were most often involved and the distal tibial metaphyses (49.6%) were the most common site.Non-steroidal anti-inflammatory drugs were used as first line (81.67%) followed by bisphosphonates (61.79%).There was no difference in number of medications used for management in unifocal versus multifocal disease.TNF blockers were used with good effect in our cohort.

show abstract

Section: Discussionmentioning

confidence: 99%

Chronic non bacterial osteitis- a multicentre study

et al. 2018

View full text Add to dashboard Cite

show abstract

“…Consensus treatment plans (CTPs) are a relatively new research methodology intended to reduce variation in treatment approaches. This process has been used for other rare diseases such as polyarticular juvenile idiopathic arthritis [11], systemic-onset juvenile idiopathic arthritis [12], juvenile dermatomyositis [13], juvenile localized scleroderma [14], chronic nonbacterial osteomyelitis [15] and juvenile lupus nephritis [16]. Therapies in use are initially identified through a literature review and physician surveys of current practice strategies.…”

Section: Introductionmentioning

confidence: 99%

Consensus treatment plans for periodic fever, aphthous stomatitis, pharyngitis and adenitis syndrome (PFAPA): a framework to evaluate treatment responses from the childhood arthritis and rheumatology research alliance (CARRA) PFAPA work group

Amarilyo

Rothman²,

Manthiram

et al. 2020

Pediatr Rheumatol

Self Cite

View full text Add to dashboard Cite

Background: Periodic fever, aphthous stomatitis, pharyngitis, and cervical adenitis (PFAPA) syndrome is the most common periodic fever syndrome in children. There is considerable heterogeneity in management strategies and a lack of evidence-based treatment guidelines. Consensus treatment plans (CTPs) are standardized treatment regimens that are derived based upon best available evidence and current treatment practices that are a way to enable comparative effectiveness studies to identify optimal therapy and are less costly to execute than randomized, double blind placebo controlled trials. The purpose of this project was to develop CTPs and response criteria for PFAPA.

show abstract

“…The difference between two groups in their response to DMARDs was likely significant due to the low rate of complete response. In the recently published consensus treatment plans, DMARDs, anti-TNF and pamidronate were listed as equivalent choices [18]. Our data suggested that both pamidronate and anti-TNF therapy were better than NSAIDs.…”

Section: Discussionmentioning

confidence: 69%

“…Treatment of CNO of the mandible is medical, and NSAIDs are generally accepted as a first-line treatment [15,17,18]. 18 of our 22 patients received NSAIDs during their treatment.…”

Section: Discussionmentioning

confidence: 99%

Pediatric chronic nonbacterial osteomyelitis of the mandible: Seattle Children’s hospital 22-patient experience

et al. 2020

Self Cite

View full text Add to dashboard Cite

Background: Studies evaluating treatment responses for chronic nonbacterial osteomyelitis (CNO) are lacking. We aimed to measure and compare response rates of medical treatments, time to response of medical treatments among patients with CNO of the mandible, and describe bacterial contamination rates from biopsy. Methods: We conducted a retrospective chart review of all patients diagnosed with CNO of mandible between 2003 and 2017 and extracted demographic, clinical, laboratory, imaging and surgical data. Detailed medication use and response to medications were recorded. The primary outcome was response to medical treatments defined as improvement of presenting symptoms, inflammatory markers, and imaging if available. Medical treatments included nonsteroidal anti-inflammatory drugs (NSAIDs), glucocorticoids, disease modifying anti rheumatic drugs (DMARDs), anti-tumor necrosis factor (TNF) therapy, and pamidronate. Descriptive analysis was performed when appropriate. Multivariable logistic regression and Kaplan-Meier curves were applied to compare the responses to medical treatments and time to full response. Results: We identified 22 patients with a median age of 11 and 36% were female. Four patients (18%) had multifocal bone lesions. CT findings (n = 21) showed lytic lesions (62%) and sclerosis (90%). MRI (n = 14) revealed hyperintensity within bone marrow (100%), soft tissue (71%) and bony expansion (71%). Non-antibiotic treatments including NSAIDs (n = 18), glucocorticoids (n = 10), DMARDs (n = 9), anti-TNF therapy (n = 5) and pamidronate (n = 6) were applied. Rates of full responses to anti-TNF therapy (60%) and pamidronate (67%) were higher than that to NSAIDs (11%) (p < 0.05). Patients receiving pamidronate responded more rapidly than those receiving anti-TNF therapy (median two vs 17 months, p = 0.01) when there was a full response. All had bone biopsies. Intraoral biopsy was performed in 12 of 13 operated in our center and the most common contaminants were Neisseria spp and Streptococcus viridians. Conclusion: Both anti-TNF and pamidronate appeared superior to NSAIDs alone in treating mandibular CNO. Patients receiving pamidronate responded faster than those receiving anti-TNF therapy.

show abstract

Consensus Treatment Plans for Chronic Nonbacterial Osteomyelitis Refractory to Nonsteroidal Antiinflammatory Drugs and/or With Active Spinal Lesions

Abstract: Three consensus treatment plans were developed for pediatric patients with CNO refractory to NSAIDs and/or with active spinal lesions. Use of these CTPs will provide additional information on efficacy and will generate meaningful data for comparative effectiveness research in CNO.

Cited by 138 publications

References 37 publications

Chronic non bacterial osteitis- a multicentre study

Chronic non bacterial osteitis- a multicentre study

Consensus treatment plans for periodic fever, aphthous stomatitis, pharyngitis and adenitis syndrome (PFAPA): a framework to evaluate treatment responses from the childhood arthritis and rheumatology research alliance (CARRA) PFAPA work group

Pediatric chronic nonbacterial osteomyelitis of the mandible: Seattle Children’s hospital 22-patient experience

Contact Info

Product

Resources

About