Cytokine profile in patients with chronic non-bacterial osteomyelitis, juvenile idiopathic arthritis, and insulin-dependent diabetes mellitus

Kostik, Mikhail M.; Makhova, Maria A.; Magomedova, Shamai; Sorokina, Lubov S.; Tsukasaki, Masayuki; Okamoto, Kazuo; Takayanagi, Hiroshi; Vasiliev, D. S.; Козлова, Д. И.; Mushkin, Aleksandr

doi:10.1016/j.cyto.2021.155521

Cited by 8 publications

(16 citation statements)

References 46 publications

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“…Although we found significantly higher levels of IL-6 and IL-8 in CNO patients with additional skin and/or joint features, these too were still markedly lower than previously reported. (9,14,30) Thus, the present study did not confirm the hypothesis of dysregulated pro-inflammatory cytokines in adult CNO. Although our study resembles previous one in terms of sample handling and the inclusion of patients with significant clinical and radiologic disease activity, several differences may explain our contrasting findings.…”

Section: Discussioncontrasting

confidence: 77%

“…2 and Table S1. ESR and CRP were higher in patients with ACW + vertebral involvement, as compared to ACW involvement alone (median [IQR] 33 versus 11 [6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21] mm/hr for ESR, p < 0.01 and 11 (5)(6)(7)(8)(9)(10)(11)(12)(13)(14) versus 3.1 (1)(2)(3)(4)(5)(6)(7) mg/L for CRP, p = 0.024, Fig. 2).…”

Section: Resultsmentioning

confidence: 99%

“…Conversely, IL‐6, IL‐8, TNF‐α, and IL‐17A were evaluated in prior studies and our findings contrast their results. IL‐6 has been found elevated in multiple pediatric CNO cohorts, ( 9,10,31 ) and also demonstrated higher concentrations in two studies on adults with SAPHO syndrome. ( 12,14 ) The same holds true for IL‐8, TNF‐α, and IL‐17A, of which the previously published means of 36.41 pg/mL, 19–25.2 pg/mL, and 29–83 pg/mL vastly exceed those found in the current study.…”

Section: Discussionmentioning

confidence: 99%

“…For adults, existing studies suggest increased expression of these cytokines too, additionally including IL-8, but data remain scarce. (9)(10)(11)(12)(13)(14) In sum, there is a need to further explore the autoinflammatory phenotype of adult CNO and assess whether it is reflected to the same degree as in children. Understanding auto-inflammatory mechanisms and key drivers of disease is of major importance to make way for rational treatments in the future.…”

Section: Introductionmentioning

confidence: 99%

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Immunological Characterization of Chronic Nonbacterial Osteomyelitis (CNO) in Adults: A Cross‐Sectional Exploratory Study

Leerling,

Andeweg,

Faber

et al. 2023

JBMR Plus

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Chronic nonbacterial osteomyelitis (CNO) is a rare disease spectrum affecting children and adults. Adult CNO may occur as isolated bone inflammation, or with a broad range of extraskeletal features. CNO pathophysiology, including the key drivers of inflammation, remains largely unknown. For pediatric CNO, a role for pro‐inflammatory cytokine dysregulation has been proposed, but studies in adults are scarce. We therefore provide immunological characterization of adult CNO. Cross‐sectional study in our referral center including adult CNO patients (n = 172) and healthy controls (n = 65). Inflammation parameters and systemic inflammatory based scores(SIBS, including neutrophil/lymphocyte ratio [NLR] and systemic immune inflammation index [SII]) were compared between groups. Cytokine expression was explored with electrochemiluminescent immunoassays in 33 patients, eight healthy controls and 21 osteoporosis patients. Routine inflammation markers were higher in patients than in controls, but generally remained within reference range. Systemic inflammation was more pronounced in patients with additional vertebral involvement as compared to those osteitis in the anterior chest wall alone, in patients with comorbid pustulosis palmoplantaris or psoriasis, and in patients with strongly rather than moderately increased lesional uptake on nuclear imaging. SII was elevated in CNO patients too, but NLR was not. Cytokine expression was generally nondifferential between patients and both control groups, and patients displayed low absolute concentrations of pro‐inflammatory cytokines. In this adult CNO cohort, systemic inflammation was generally subtle, but more pronounced in patients with vertebral lesions, associated skin disease, and strongly increased uptake on nuclear imaging. SII was increased in patients compared to healthy controls. Contrasting pediatric studies, we found no increased expression of the pro‐inflammatory cytokines that have been proposed to drive the inflammatory cascade, like interleukin‐6, ‐8, and ‐17 (IL‐6, IL‐8, and IL‐17), and tumor necrosis α (TNF‐α). Further studies are needed to evaluate the use of SII in diagnosis and monitoring of CNO, and elucidate the role of cytokine dysregulation in adult disease. © 2023 The Authors. JBMR Plus published by Wiley Periodicals LLC. on behalf of American Society for Bone and Mineral Research.

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Section: Discussioncontrasting

confidence: 77%

Section: Resultsmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Immunological Characterization of Chronic Nonbacterial Osteomyelitis (CNO) in Adults: A Cross‐Sectional Exploratory Study

Leerling,

Andeweg,

Faber

et al. 2023

JBMR Plus

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“…Recently, some authors have discussed the role of serum cytokines and chemokines, such as S100A8/A9, in differentiating CNO from other conditions such as leukemia, infections, IBD, and healthy control [ 26 , 27 ]. More studies are required to evaluate the use of these biomarkers in the process of diagnosis, treatment strategy, and follow-up in CNO patients.…”

Section: Laboratory Findingsmentioning

confidence: 99%

Chronic Nonbacterial Osteomyelitis in Children

et al. 2021

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Chronic nonbacterial osteomyelitis (CNO) is an auto-inflammatory bone disorder with a wide spectrum of clinical manifestations, from unifocal to multifocal lesions. When it manifests with multifocal lesions, it is also referred to as chronic recurrent multifocal osteomyelitis (CRMO). CNO/CRMO can affect all age groups, with the pediatric population being the most common. Patients may present with systemic inflammation, but there is no pathognomonic laboratory finding. Magnetic resonance imaging (MRI) is the gold standard radiological tool for diagnosis. In the absence of validated diagnostic criteria, CNO/CRMO remains an exclusion diagnosis. Bone biopsy does not show a specific disease pattern, but it may be necessary in unifocal or atypical cases to differentiate it from malignancy or infection. First-line treatments are non-steroidal anti-inflammatory drugs (NSAIDs), while bisphosphonates or TNF-α blockers can be used in refractory cases. The disease course is unpredictable, and uncontrolled lesions can complicate with bone fractures and deformations, underlying the importance of long-term follow-up in these patients.

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Asocieri rare în artrita juvenilă idiopatică – osteomielita cronică nonbacteriană

Murgu¹,

Macsim²,

Apostol³

et al. 2023

Pediatru.ro

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Cytokine profile in patients with chronic non-bacterial osteomyelitis, juvenile idiopathic arthritis, and insulin-dependent diabetes mellitus

Cited by 8 publications

References 46 publications

Immunological Characterization of Chronic Nonbacterial Osteomyelitis (CNO) in Adults: A Cross‐Sectional Exploratory Study

Immunological Characterization of Chronic Nonbacterial Osteomyelitis (CNO) in Adults: A Cross‐Sectional Exploratory Study

Chronic Nonbacterial Osteomyelitis in Children

Asocieri rare în artrita juvenilă idiopatică – osteomielita cronică nonbacteriană

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