Classification and management strategies for paediatric chronic nonbacterial osteomyelitis and chronic recurrent multifocal osteomyelitis

Singhal, Shabnam; Landes, Caren; Shukla, Rajeev; McCann, Liza; Hedrich, Christian M.

doi:10.1080/1744666x.2023.2218088

Cited by 7 publications

(5 citation statements)

References 96 publications

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“… 56 Skin disease, on the other hand, may be the result of adaptive immune dysregulation and the differentiation and activation of effector T cell phenotypes. 39 43 …”

Section: Discussionmentioning

confidence: 99%

“…Most studies addressing disease mechanisms focus on paediatric CNO/CRMO. [39][40][41] Because of clinical overlaps, some authors suggested that SAPHO may be a 'late stage' or the adult form of CNO/CRMO, and the result of ongoing innate immune activation that, over time, may result in the activation of adaptive immune mechanisms and pathological activation of effector T lymphocytes. Adaptive immune activation may, through the release of effector T cell-derived cytokines (such as IL-12, IL-17A, IL-23), result in the development of skin and nail changes frequently seen in SAPHO.…”

Section: Treatments Treatments Treatmentsmentioning

confidence: 99%

“…Methotrexate (MTX) and sulfasalazine (SSZ) reduce proinflammatory cytokine expression. 39 43 56 NSAIDs, non-steroidal anti-inflammatory drugs; SAPHO, synovitis acne pustulosis hyperostosis osteitis.…”

Section: Introductionmentioning

confidence: 99%

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Treatment and monitoring of SAPHO syndrome: a systematic review

Li,

Roberts,

Hedrich

2023

RMD Open

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Background and objectivesSynovitis acne pustulosis hyperostosis osteitis (SAPHO) is a rare heterogeneous disease of unknown aetiopathology. Externally validated and internationally agreed diagnostic criteria or outcomes and, as a result, prospective randomised controlled trials in SAPHO are absent. Consequently, there is no agreed treatment standard. This study aimed to systematically collate and discuss treatment options in SAPHO.MethodsFollowing ‘Preferred Reporting Items for Systematic Reviews and Meta-Analyses’ guidance, a systematic literature search was conducted using PubMed, Scopus and Web of Science databases. Prospective clinical studies and retrospective case collections discussing management and outcomes in SAPHO involving five or more participants were included. Articles not published in English, studies not reporting defined outcomes, and studies solely relying on patient-reported outcomes were excluded.ResultsA total of 28 studies (20 observational, 8 open-label clinical studies) reporting 796 patients of predominantly European ethnicity were included. Reported therapies varied greatly, with many centres using multiple treatments in parallel. Most patients (37.1%) received non-steroidal anti-inflammatory drugs alone or in combination. Bisphosphonates (22.1%), conventional (21.7%) and biological (11.3%) disease-modifying antirheumatic drugs were the next most frequently reported treatments. Reported outcomes varied and delivered mixed results, which complicates comparisons. Bisphosphonates demonstrated the most consistent improvement of osteoarticular symptoms and were associated with transient influenza-like symptoms. Paradoxical skin reactions were reported in patients treated with TNF inhibitors, but no serious adverse events were recorded. Most treatments had limited or mixed effects on cutaneous involvement. A recent study investigating the Janus kinase inhibitor tofacitinib delivered promising results in relation to skin and nail involvement.ConclusionsNo single currently available treatment option sufficiently addresses all SAPHO-associated symptoms. Variable, sometimes descriptive outcomes and the use of treatment combinations complicate conclusions and treatment recommendations. Randomised clinical trials are necessary to generate reliable evidence.

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“… 56 Skin disease, on the other hand, may be the result of adaptive immune dysregulation and the differentiation and activation of effector T cell phenotypes. 39 43 …”

Section: Discussionmentioning

confidence: 99%

Section: Treatments Treatments Treatmentsmentioning

confidence: 99%

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Treatment and monitoring of SAPHO syndrome: a systematic review

Li,

Roberts,

Hedrich

2023

RMD Open

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“…Other authors have proposed diagnostic criteria and biomarkers for CNO, but these have not been validated in prospective studies, and, to date, CNO/CRMO remains a diagnosis of exclusion [9,10].…”

Section: Introductionmentioning

confidence: 99%

“…Regarding the treatment of CNO in one of the most frequent locations, the clavicular region (about 5% of cases [11]), the literature reports different treatment options. Observation, medical therapy with antibiotics, non-steroidal anti-inflammatory drugs (NSAIDs) or bisphosphonates, TNF inhibitors, disease-modifying antirheumatic drugs (DMARDS), [10,[12][13][14][15][16] and surgical excision of the lesion. The surgical treatment for CNO of the clavicle remains debated among intralesional curettage, partial resection, and total clavicular resection (TCR) [17][18][19].…”

Section: Introductionmentioning

confidence: 99%

Garrè Sclerosing Osteomyelitis of the Clavicle: Clinical Results after Clavicular Resection

Calamita,

Stimolo,

Puccini

et al. 2024

Healthcare

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(1) Background: Chronic non-bacterial osteomyelitis (CNO), also known as sclerosing osteomyelitis of Garrè, is a rare inflammatory bone disease with a specific clinical picture, uncertain pathogenesis, and no consensus on an effective treatment. Most frequently affecting other long bones, CNO may rarely involve the clavicle. The aim of this study was to present the results of a series of patients affected by CNO of the clavicle treated with total and partial clavicula resection. In addition, a literature review of different types of treatment of CNO was performed. (2) Methods: We retrospectively reviewed three patients with Sclerosing Osteomyelitis of Garre’ of the clavicle treated with partial resection of the clavicle (one) and with total clavicular resection (two). (3) Results: Patients (two female and one male) were an average age of 35.7 years at the time of the operation. At the 4-year follow-up, the mean active ROM was: 143° forward flexion, 133° abduction, 42° external rotation with an internal rotation of two patients at the interscapular level and one patient at the lumbosacral junction. The mean ASES score was 92/100 (range 87–100). In the literature review, after screening the abstracts and full texts for eligibility, 34 studies met the inclusion criteria. Conclusions: Partial or total clavicular resection resulted an effective treatment of CNO of the clavicle. The procedure seems to be particularly indicated after the failure of more conservative treatments.

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