TNF-inhibitors or bisphosphonates in chronic nonbacterial osteomyelitis? - Results of an international retrospective multicenter study

Schnabel, Anja; Nashawi, Mohammed; Anderson, Catriona M.H.; Felsenstein, Susanna; Lamoudi, Maya; Poole-Cowley, J.; Lindell, E.; Fowlie, Peter W; Walsh, Jo; Ellis, T.; Hahn, Gabriele; Goldspink, A.; Martin, Neil; Mahmood, Kamran; Hospach, Toni; McCann, Liza; Hedrich, Christian M.

doi:10.1016/j.clim.2022.109018

Cited by 21 publications

(25 citation statements)

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“…In cases of more severe CRMO, instead of first-line medicaments, NSAIDs or, in combination with them, a wide range of second-line treatment options is available, including disease-modifying anti-rheumatic drugs (DMARDs), glucocorticoids and bisphosphonates [ 25 , 26 ]. Pamidronate, classified as a nitrogen-containing bisphosphonate, inhibits farnesyl diphosphate synthase in osteoclasts by mimicking its substrate.…”

Section: Discussionmentioning

confidence: 99%

“…As for therapeutic safety, its efficacy was also ambiguously discussed in recent studies. Some reports state that more than 2/3 patients treated with bisphosphonates achieved remission of CRMO symptoms and the progression of their disorder was inhibited, both in spinal and peripheral lesions [ 25 , 26 , 29 , 30 , 31 , 32 , 33 , 34 ]. It was also suggested that pamidronate leads to a rapid improvement in patient’s condition [ 26 ], which is in accordance with the observations of this study—within 1.5 years the reported patient returned to regular height range (50.-75. centile) from the threshold of 10. centile.…”

Section: Discussionmentioning

confidence: 99%

“…Some reports state that more than 2/3 patients treated with bisphosphonates achieved remission of CRMO symptoms and the progression of their disorder was inhibited, both in spinal and peripheral lesions [ 25 , 26 , 29 , 30 , 31 , 32 , 33 , 34 ]. It was also suggested that pamidronate leads to a rapid improvement in patient’s condition [ 26 ], which is in accordance with the observations of this study—within 1.5 years the reported patient returned to regular height range (50.-75. centile) from the threshold of 10. centile. It should also be noted, that pamidronate was used in this case as a monotherapy, as opposed to previous reports where the preferred method was combination with DMARDs or glucocorticoids [ 35 ].…”

Section: Discussionmentioning

confidence: 99%

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A Successful Bisphosphonates Monotherapy in Spinal Form of Paediatric Chronic Recurrent Multifocal Osteomyelitis (CRMO)—Case Report

et al. 2023

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Chronic recurrent multifocal osteomyelitis (CRMO) is a non-infectious inflammatory disorder resulting from the multifocal bone and bone marrow lesions with periodic relapses and remissions and with an uncertain prognosis. Treatment options in CRMO are based on expert opinion and relatively small groups of patients. A nine-year-old female patient with no significant past medical history presented with compression fractures and multifocal bone lesions in the thoracic and lumbar spine, as shown in imaging (CT, MRI). Densitometry revealed a diffuse decrease in bone density. Based on the patient’s clinical image and above examinations, the other possible aetiologies—infectious (including tuberculosis), neoplasms, Langerhans cell histiocytosis—were ruled out, which led to eventual final diagnosis—CRMO. The patient was successfully treated with pamidronate infusion initiated in cycles over three consecutive days every 3 months. In addition to clinical improvement, there was a significant remission of inflammation and bone structure healing assessed by MRI after four treatment cycles. Intravenous bisphosphonates usage seems to be a good therapeutic option in CRMO paediatric patients with spinal localization of the lesions complicated by compressive fractures. However, more data, based on larger patient populations, are needed to provide a detailed paediatric CRMO treatment algorithm.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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A Successful Bisphosphonates Monotherapy in Spinal Form of Paediatric Chronic Recurrent Multifocal Osteomyelitis (CRMO)—Case Report

et al. 2023

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“…Studies have shown that serum pro-inflammatory molecules (IL-6, TNF-α, IL-1β) are increased and anti-inflammatory factors (IL-10, IL-19) are decreased in CNO patients ( 29 ). The imbalance between pro- and anti-inflammatory cytokines can be restored by TNF-α inhibitors (TNFi) which are recommended as the preferred second-line treatment for patients with associated inflammatory skin lesions ( 30 ). Our patient had pustular lesions on the lower limbs, so we added the TNFi adalimumab.…”

Section: Discussionmentioning

confidence: 99%

Case report: Child chronic nonbacterial osteomyelitis with rapid progressive scoliosis-an association with disease?

et al. 2023

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BackgroundChronic nonbacterial osteomyelitis (CNO) is an auto-inflammatory bone disease that usually develops in childhood. Spinal involvement is a common manifestation of CNO, but it is rare for CNO to lead to rapid progression of scoliosis deformity. Here we present a 9-year-old girl with acute scoliosis with CNO and scoliosis progressed rapidly in 2 months.Case PresentationA 9-year-old girl presented bilateral shoulder inequality with pain in the left hypochondrium for 2 months. Standing spinal x-rays showed right convex scoliosis with a 25° Cobb angle. Chest magnetic resonance imaging (MRI) showed that the T8 vertebra was flattened and local bone was destroyed with bone marrow edema. The bone biopsy showed evidence of fibrosis and chronic inflammatory changes with no specific diagnosis. One month later, her scoliosis and bone destruction deteriorated obviously. Thoracic vertebra MRI showed that the T8 vertebra had a compression fracture. 99mTc-MDP whole-body bone scintigraphy showed intense uptake at T8/9 and the right sacroiliac joint. She was diagnosed with CNO accompanied by rapidly progressive scoliosis. The scoliosis was successfully treated with adalimumab and zoledronic acid, which showed significant improvement after 6 months of follow-up.ConclusionZoledronic acid and adalimumab successfully treated CNO with rapidly progressive scoliosis, but could not prevent vertebral compression.

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“…Recently, a retrospective chart review from Germany and the UK reported that both TNFi and pamidronate were safe and effective in patients with CNO who failed NSAID treatment (13). There appeared to be a quicker treatment response in patients treated with pamidronate, but more sustained remission in the TNFi group (13). Due to lack of robust evidence from prospective international and multi-ethnic studies, supporting the therapeutic efficacy and safety of any of the second-line treatments, the Childhood Arthritis and Rheumatology Research Alliance (CARRA) CNO working group adopted all three categories of treatments as consensus treatment plans (CTPs) for children with CNO who had an NSAID-refractory course and/or with active spinal lesions (14).…”

Section: Introductionmentioning

confidence: 99%

Feasibility of Conducting Comparative Effectiveness Research and Validation of a Clinical Disease Activity Score for Chronic Nonbacterial Osteomyelitis

Wu¹,

Oliver²,

Scheck³

et al. 2022

Preprint

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Objectives: Study objectives were to: 1) determine the frequency of use and safety of each consensus treatment plan (CTP) regimen for chronic nonbacterial osteomyelitis (CNO) and the feasibility of using chronic nonbacterial osteomyelitis international registry (CHOIR) data for comparative effectiveness research and 2) develop and validate a CNO clinical disease activity score (CDAS) using CHOIR. Methods: Consenting CNO children or young adults were enrolled into CHOIR. Demographic, clinical, and imaging data were prospectively collected. The CNO CDAS was developed through a Delphi survey and nominal group technique. External validation surveys were administered to CHOIR participants. Results: 140 (76%) CHOIR participants enrolled between August 2018 and September 2020 received at least one CTP regimen. Baseline characteristics from the three groups were well-matched. Patient pain, patient global assessment, and clinical CNO lesion count were key variables included in the CNO CDAS. The CDAS showed a strong correlation with patient/parent report of difficulty using a limb, back, or jaw and patient/parent report of disease severity, but a weak correlation with patient/parent report of fatigue, sadness, and worry. The change in CDAS was significant in patients reporting disease worsening or improvement. The CDAS significantly decreased after initiating second-line treatments from 12 (8-15.5) to 5 (3-12). All second-line treatments were well-tolerated. Psoriasis was the most common adverse event. Conclusion: The CNO CDAS was developed and validated for disease monitoring and assessment of treatment effectiveness. CHOIR provided a comprehensive framework for future comparative effectiveness research.

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TNF-inhibitors or bisphosphonates in chronic nonbacterial osteomyelitis? - Results of an international retrospective multicenter study

Cited by 21 publications

References 62 publications

A Successful Bisphosphonates Monotherapy in Spinal Form of Paediatric Chronic Recurrent Multifocal Osteomyelitis (CRMO)—Case Report

A Successful Bisphosphonates Monotherapy in Spinal Form of Paediatric Chronic Recurrent Multifocal Osteomyelitis (CRMO)—Case Report

Case report: Child chronic nonbacterial osteomyelitis with rapid progressive scoliosis-an association with disease?

Feasibility of Conducting Comparative Effectiveness Research and Validation of a Clinical Disease Activity Score for Chronic Nonbacterial Osteomyelitis

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