Plasma Levels of Leptin and Adiponectin in Fragile X Syndrome

Lisik, Małgorzata; Gutmajster, Ewa; Sieroń, Aleksander

doi:10.1159/000452336

Cited by 16 publications

(18 citation statements)

References 28 publications

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“…Consistent with these findings, Rodrigues et al, noted that adiponectin levels were inversely correlated with the severity of autism symptoms (Rodrigues et al 2014). Lisik et al showed that adiponectin levels were lower in those with Fragile X Syndrome (Lisik et al 2016). These findings suggest that adiponectin may have a larger role to play in neurodevelopmental processes, rather than merely regulate energy expenditure or serve as a biomarker for the onset of metabolic syndrome (Inami et al 2007; Blardi et al 2009).…”

Section: Discussionmentioning

confidence: 99%

“…First, as an anti-inflammatory cytokine, adiponectin functions as a mediator of inflammatory response and has a protective role against metabolic disturbances (Mazaki-Tovi et al 2009; Lenz and Diamond 2012; Fujita-Shimizu et al 2010). Emerging evidence suggests that adiponectin may be implicated in neurological conditions (Lisik et al 2016; Mansur et al 2016; Hu et al 2015) (Burd et al 2012). To our knowledge, three studies have assessed adiponectin levels in children with ASD (Fujita-Shimizu et al 2010; Blardi et al 2010; Rodrigues et al 2014), of which only one found altered adiponectin levels in subjects with ASD (Fujita-Shimizu et al 2010).…”

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confidence: 99%

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Cord and Early Childhood Plasma Adiponectin Levels and Autism Risk: A Prospective Birth Cohort Study

Raghavan

Fallin

Hong

et al. 2018

J Autism Dev Disord

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Emerging research suggests that adiponectin, a cytokine produced by adipose tissue, may be implicated in ASD. In this prospective birth cohort study (n = 847), we assessed the association between cord, early childhood plasma adiponectin and the risk of developing ASD. ASD was defined based on ICD codes of physician diagnosis. Cord adiponectin levels were inversely associated with ASD risk (aOR 0.50; 95% CI 0.33, 0.77), independent of preterm birth, early childhood adiponectin and other known ASD risk factors. Early childhood adiponectin, assessed prior to ASD diagnosis, was associated with lower risk of ASD, which attenuated after adjusting for cord adiponectin, indicating the relative importance of cord adiponectin in ASD risk. Further research is warranted to confirm our findings and elucidate biological mechanisms.

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Section: Discussionmentioning

confidence: 99%

mentioning

confidence: 99%

Cord and Early Childhood Plasma Adiponectin Levels and Autism Risk: A Prospective Birth Cohort Study

Raghavan

Fallin

Hong

et al. 2018

J Autism Dev Disord

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“…To study possible peripheral consequences of FMR1deficiency, some authors have compared the levels of serum and plasma biomarkers between FXS patients and healthy controls. Prior studies have focused on metabolic markers and have highlighted reduced levels of cholesterol [11][12][13] and abnormal abundances of the metabolic hormones leptin and adiponectin [14]. In a translational study addressing metabolic consequences of FMR1-deficiency both in a FXS mouse model and in FXS patients, we have recently shown that FXS patients display reductions in circulating glucose and increases in both free fatty acids and insulin, underlining metabolic anomalies in FXS [15].…”

Section: Intellectual Disabilitymentioning

confidence: 99%

Reduced serum levels of pro-inflammatory chemokines in fragile X syndrome

et al. 2020

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Background: Fragile X syndrome (FXS) is the most frequent cause of inherited intellectual disability and the most commonly identified monogenic cause of autism. Recent studies have shown that long-term pathological consequences of FXS are not solely confined to the central nervous system (CNS) but rather extend to other physiological dysfunctions in peripheral organs. To gain insights into possible immune dysfunctions in FXS, we profiled a large panel of immune-related biomarkers in the serum of FXS patients and healthy controls. Methods: We have used a sensitive and robust Electro Chemi Luminescence (ECL)-based immunoassay to measure the levels of 52 cytokines in the serum of n = 25 FXS patients and n = 29 healthy controls. We then used univariate statistics and multivariate analysis, as well as an advanced unsupervised clustering method, to identify combinations of immune-related biomarkers that could discriminate FXS patients from healthy individuals. Results: While the majority of the tested cytokines were present at similar levels in FXS patients and healthy individuals, nine chemokines, CCL2, CCL3, CCL4, CCL11, CCL13, CCL17, CCL22, CCL26 and CXCL10, were present at much lower levels in FXS patients. Using robust regression, we show that six of these biomarkers (CCL2, CCL3, CCL11, CCL22, CCL26 and CXCL10) were negatively associated with FXS diagnosis. Finally, applying the K-sparse unsupervised clustering method to the biomarker dataset allowed for the identification of two subsets of individuals, which essentially matched the FXS and healthy control categories. Conclusions: Our data show that FXS patients exhibit reduced serum levels of several chemokines and may therefore exhibit impaired immune responses. The present study also highlights the power of unsupervised clustering methods to identify combinations of biomarkers for diagnosis and prognosis in medicine.

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“…To study possible peripheral consequences of FMR1-deficiency, some authors have compared the levels of serum and plasma biomarkers between FXS patients and healthy controls. Prior studies have focused on metabolic markers and have highlighted reduced levels of cholesterol [11][12][13] and abnormal abundances of the metabolic hormones leptin and adiponectin [14]. In a translational study addressing metabolic consequences of FMR1-deficiency both in a FXS mouse model and in FXS patients, we have recently shown that FXS patients display reductions in circulating glucose and increases in both free fatty acids and insulin, underlining metabolic anomalies in FXS [15].…”

Section: Introductionmentioning

confidence: 99%

Reduced serum levels of pro-inflammatory chemokines in Fragile X Syndrome

Dijck

Barbosa

Bermudez-Martin

et al. 2020

Preprint

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Background: Fragile X syndrome (FXS) is the most frequent cause of inherited intellectual disability and the most commonly identified monogenic cause of autism. Recent studies have shown that long-term pathological consequences of FXS are not solely confined to the central nervous system (CNS) but rather extend to other physiological dysfunctions in peripheral organs. To gain insights into possible immune dysfunctions in FXS, we profiled a large panel of immune-related biomarkers in the serum of FXS patients and healthy controls. Methods: We have used a sensitive and robust Electro Chemi Luminescence (ECL)-based immunoassay to measure the levels of 52 cytokines in the serum of n=25 FXS patients and n=29 healthy controls. We then used univariate statistics and multivariate analysis, as well as an advanced unsupervised clustering method, to identify combinations of immune-related biomarkers that could discriminate FXS patients from healthy individuals. Results: While the majority of the tested cytokines were present at similar levels in FXS patients and healthy individuals, nine chemokines, CCL2, CCL3, CCL4, CCL11, CCL13, CCL17, CCL22, CCL26 and CXCL10, were present at much lower levels in FXS patients. Using robust regression, we show that six of these biomarkers (CCL2, CCL3, CCL11, CCL22, CCL26 and CXCL10) were negatively associated with FXS diagnosis. Finally, applying the K-sparse unsupervised clustering method to the biomarker dataset allowed for the identification of two subsets of individuals, which essentially matched the FXS and healthy control categories. Conclusions: Our data show that FXS patients exhibit reduced serum levels of several chemokines. This paves the way for further study of immune phenotypes in FXS patients.

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Plasma Levels of Leptin and Adiponectin in Fragile X Syndrome

Cited by 16 publications

References 28 publications

Cord and Early Childhood Plasma Adiponectin Levels and Autism Risk: A Prospective Birth Cohort Study

Cord and Early Childhood Plasma Adiponectin Levels and Autism Risk: A Prospective Birth Cohort Study

Reduced serum levels of pro-inflammatory chemokines in fragile X syndrome

Reduced serum levels of pro-inflammatory chemokines in Fragile X Syndrome

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