Candidiasis associated with very early onset inflammatory bowel disease: First IL10RB deficient case from the National Iranian Registry and review of the literature

Yazdani, Reza; Moazzami, Bobak; Madani, Seyedeh Panid; Behniafard, Nasrin; Azizi, Gholamreza; Aflatoonian, Majid; Abolhassani, Hassan; Aghamohammadi, Asghar

doi:10.1016/j.clim.2019.05.007

Cited by 10 publications

(9 citation statements)

References 40 publications

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“…IL-10 was also related with greater inflammatory markers in the sepsis group. According to the research, IL-10 is produced by a variety of cells and has a crucial function in limiting the production of proinflammatory cytokines, such as IL-12, which are generated when a person is inflamed (26). In the infected group, there was a positive correlation between IL-17 levels and IL-8, but in the sepsis group, there was a positive correlation between IL-6 and IL-8, showing that IL-17 is better able to react to inflammation markers.…”

Section: Discussionmentioning

confidence: 99%

The diagnostic utility of IL-10, IL-17, and PCT in patients with sepsis infection

Zhang

Wang

Hou

et al. 2022

Front. Public Health

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ObjectiveThe purpose of this study is to determine the diagnostic value and net clinical benefit of interleukin-10 (IL-10), interleukin-17 (IL-17), procalcitonin (PCT), and combination tests in patients with sepsis, which will serve as a standard for sepsis early detection.Patients and methodsAn investigation of 84 sepsis patients and 81 patients with local inflammatory diseases admitted to the ICU of Tongji University Hospital in 2021. In addition to comparing inter-group variability, indicators relevant to sepsis diagnosis and therapy were screened.ResultsLASSO regression was used to examine PCT, WBC, CRP, IL-10, IFN-, IL-12, and IL-17. Multivariate logistic regression linked IL-10, IL-17, and PCT to sepsis risk. The AUC values of IL-10, IL-17, PCT, and the combination of the three tests were much higher than those of standard laboratory infection indicators. The combined AUC was greater than the sum of IL-10, IL-17, and PCT (P < 0.05). A clinical decision curve analysis of IL-10, IL-17, PCT, and the three combined tests found that the three combined tests outperformed the individual tests in terms of total clinical benefit rate. To predict the risk of sepsis using IL-10, IL-17, and PCT had an AUC of 0.951, and the model's predicted probability was well matched. An examination of the nomogram model's clinical value demonstrated a considerable net therapeutic benefit between 3 and 87%.ConclusionThe IL-10, IL-17, and PCT tests all have a high diagnostic value for patients with sepsis, and the combination of the three tests outperforms the individual tests in terms of diagnostic performance, while the combined tests have a higher overall clinical benefit rate.

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Section: Discussionmentioning

confidence: 99%

The diagnostic utility of IL-10, IL-17, and PCT in patients with sepsis infection

Zhang

Wang

Hou

et al. 2022

Front. Public Health

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“…These clinical observations are consistent with the incomplete blockade of IL-17 receptor signaling conferred by these monoclonal antibodies at the mucocutaneous barrier (116,117), as opposed to the complete abrogation of IL-17 receptor signaling in patients with inherited complete deficiencies of the IL-17 receptors IL-17RA and IL-17RC and of the IL-17 receptor adaptor ACT1 who develop CMC with complete penetrance (118)(119)(120). Furthermore, patients with inherited IL-10RB deficiency whose cells do not respond to IL-22 (nor to IL-10, IL-26, IL-28, and IFNL1) do not develop CMC; a single case of treatment-responsive OPC in the absence of iatrogenic immunosuppression has been reported in IL-10RB-deficient patients (OPC frequency, ∼3%) who develop very severe early-onset inflammatory bowel disease requiring treatment with corticosteroids and/or TNF-α inhibitors (121)(122)(123). Taken together, although APECED patients harbor neutralizing autoantibodies against type-17 cytokines, their presence is unlikely to be the sole factor that might contribute to CMC in the APECED population (124).…”

Section: Autoantibodies Against Type-17 Cytokines Ifn-γ-driven Defects In Oral Epithelial Barrier and Cmcmentioning

confidence: 99%

Autoimmune Polyendocrinopathy-Candidiasis-Ectodermal Dystrophy

2021

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Autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy (APECED), also known as autoimmune polyglandular syndrome type-1 (APS-1), is a rare monogenic autoimmune disease caused by loss-of-function mutations in the autoimmune regulator (AIRE) gene. AIRE deficiency impairs immune tolerance in the thymus and results in the peripheral escape of self-reactive T lymphocytes and the generation of several cytokine- and tissue antigen-targeted autoantibodies. APECED features a classic triad of characteristic clinical manifestations consisting of chronic mucocutaneous candidiasis (CMC), hypoparathyroidism, and primary adrenal insufficiency (Addison's disease). In addition, APECED patients develop several non-endocrine autoimmune manifestations with variable frequencies, whose recognition by pediatricians should facilitate an earlier diagnosis and allow for the prompt implementation of targeted screening, preventive, and therapeutic strategies. This review summarizes our current understanding of the genetic, immunological, clinical, diagnostic, and treatment features of APECED.

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“…However, the impact of pathogenic IL10RB variants on IL-10RB-dependent cytokine response pathways has not been studied experimentally. Predicted loss-of-function (pLOF) variants of IL10RB have been reported to cause a premature termination of translation in various domains of the protein, whereas missense pathogenic mutations are clustered in the extracellular domain [44,48,50,52,[56][57][58] (Fig. 1D).…”

Section: Impaired Production and Glycosylation Of The W100g Proteinmentioning

confidence: 99%

Fulminant Viral Hepatitis in Two Siblings with Inherited IL-10RB Deficiency

et al. 2022

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Fulminant viral hepatitis (FVH) caused by hepatitis A virus (HAV) is a life-threatening disease that typically strikes otherwise healthy individuals. The only known genetic etiology of FVH is inherited IL-18BP deficiency, which unleashes IL-18-dependent lymphocyte cytotoxicity and IFN-γ production. We studied two siblings who died from a combination of early-onset inflammatory bowel disease (EOIBD) and FVH due to HAV. The sibling tested was homozygous for the W100G variant of IL10RB previously described in an unrelated patient with EOIBD. We show here that the out-of-frame IL10RB variants seen in other EOIBD patients disrupt cellular responses to IL-10, IL-22, IL-26, and IFN-λs in overexpression conditions and in homozygous cells. By contrast, the impact of in-frame disease-causing variants varies between cases. When overexpressed, the W100G variant impairs cellular responses to IL-10, but not to IL-22, IL-26, or IFN-λ1, whereas cells homozygous for W100G do not respond to IL-10, IL-22, IL-26, or IFN-λ1. As IL-10 is a potent antagonist of IFN-γ in phagocytes, these findings suggest that the molecular basis of FVH in patients with IL-18BP or IL-10RB deficiency may involve excessive IFN-γ activity during HAV infections of the liver. Inherited IL-10RB deficiency, and possibly inherited IL-10 and IL-10RA deficiencies, confer a predisposition to FVH, and patients with these deficiencies should be vaccinated against HAV and other liver-tropic viruses.

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Candidiasis associated with very early onset inflammatory bowel disease: First IL10RB deficient case from the National Iranian Registry and review of the literature

Cited by 10 publications

References 40 publications

The diagnostic utility of IL-10, IL-17, and PCT in patients with sepsis infection

The diagnostic utility of IL-10, IL-17, and PCT in patients with sepsis infection

Autoimmune Polyendocrinopathy-Candidiasis-Ectodermal Dystrophy

Fulminant Viral Hepatitis in Two Siblings with Inherited IL-10RB Deficiency

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