Comparison of the serum cytokine levels before and after adrenocorticotropic hormone (ACTH) therapy in patients with infantile spasm

Türe, Esra; Kamaşak, Tülay; Cora, Merve; Şahin, Sevim; Arslan, Elif Acar; Kaklıkaya, Neşe; Cansu, Ali

doi:10.1016/j.seizure.2016.06.021

Cited by 12 publications

(13 citation statements)

References 20 publications

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“…[2,10] IL-2 and TNF-α are the main cytokines that are involved in inflammation and have recently been associated with infantile spasm. [3] Liu et al [6] revealed that the expression levels of IL-2 are higher in patients with WS than in controls. IL-2 is secreted by activated CD4 + T cells and CD8 + T cells.…”

Section: Discussionmentioning

confidence: 99%

“…[2] Previous studies have shown that immunomodulatory dysfunction in humoral immunology and cytokine levels in patients with WS. [3] Patients with the clinical feature of epileptic spasms have higher levels of antibodies against the N-methyl-d-aspartate-type glutamate receptor in the cerebrospinal fluid. [4] The levels of serum immunoglobulins (Igs) and complement proteins of IgA, IgM, IgG, C3, and C4 have been found to be lower in patients with WS than in control patients.…”

Section: Introductionmentioning

confidence: 99%

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Spontaneous remission of West syndrome following a human herpesvirus 7 infection in a Chinese infant

Wang²,

Shan³

et al. 2019

Medicine

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Rationale: West syndrome (WS) is an age-dependent epileptic encephalopathy that is characterized by intractable epileptic seizures, hypsarrhythmia, and observed through electroencephalogram (EEG) and significant neurodevelopmental regression. The spontaneous remission of epileptic seizure is clinically rare and has not previously been reported in a Chinese infant. Herein, we reported a Chinese infant with WS whose seizures disappeared following a human herpesvirus 7 (HHV-7) infection. Patient concerns: The male Chinese infant was born at the gestational age of 36 weeks with a birth weight of 1.65 kg and an Apgar score of 7 at the first minute. At the age of 6 months, the infant developed seizures that manifested as flexor spasms with trunk involvement and mental regression. Diagnosis: Brain magnetic resonance imaging revealed leukomalacia of the posterior horn and a reduction in the size of the periventricular of the bilateral ventricle and the corpus callosum. An EEG revealed hypsarrhythmia and typical spasm seizures. Therefore, the infant was diagnosed with symptomatic WS. Interventions: The infant was treated with adequate vitamin B6 intravenous drip and oral treatment with topiramate and levetiracetam. Outcomes: The observed seizures disappeared spontaneously 40 days after onset, without any changes in the anti-epileptic drug treatment, following a febrile rash due to a HHV-7 infection. Lessons: Spontaneous remission of epileptic seizures can occur following viral infection of HHV-7 in children with WS. The mechanism behind this spontaneous remission warrants further research.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Spontaneous remission of West syndrome following a human herpesvirus 7 infection in a Chinese infant

Wang²,

Shan³

et al. 2019

Medicine

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“…Here we show that the complement C5ar1 receptor antagonist, PMX53, also reduced spasms, suggesting contribution of the innate inflammatory system, represented by C5a, to IS. The involvement of the inflammatory system in IS was recently documented by the increase of cytokines IL6 and IL17A in patients with IS prior to treatment initiation compared to healthy individuals 47 , 48 .…”

Section: Discussionmentioning

confidence: 99%

ACTH and PMX53 recover synaptic transcriptome alterations in a rat model of infantile spasms

Iacobaş

Chachua

Iacobaş

et al. 2018

Sci Rep

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We profiled the gene expression in the hypothalamic arcuate nuclei (ARC) of 20 male and 20 female rats to determine the infantile spasms (IS) related transcriptomic alteration of neurotransmission and recovery following two treatments. Rats were prenatally exposed to betamethasone or saline followed by repeated postnatal subjection to NMDA-triggered IS. Rats with spasms were treated with ACTH, PMX53 or saline. Since ACTH, the first line treatment for IS, has inconsistent efficacy and potential harsh side effects, PMX53, a potent complement C5ar1 antagonist, was suggested as a therapeutic alternative given its effects in other epilepsy models. Novel measures that consider all genes and are not affected by arbitrary cut-offs were used, in addition to standard statistical tests, to quantify regulation and recovery of glutamatergic, GABAergic, cholinergic, dopaminergic and serotonergic pathways. Although IS alters expression of ~30% of the ARC genes in both sexes the transcriptomic effects are 3× more severe in males than their female counterparts, as indicated by the Weighted Pathway Regulation measure. Both treatments significantly restored the ARC neurotransmission transcriptome to the non-IS condition with PMX53 performing slightly better, as measured by the Pathway Restoration Efficiency, suggesting these treatments may reduce autistic traits often associated with IS.

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“…Although these levels decreased after ACTH treatment, the change was not significant. 12 In our previous study, post-treatment blood sampling was performed within 1 week after symptom improvement. However, Shiihara and Türe performed blood sampling after 1 month.…”

Section: Discussionmentioning

confidence: 99%

“…Comparative analyses of cytokine levels in West syndrome patients before and after ACTH treatment are limited. [10][11][12] To our knowledge, a serial analysis of the responses of multiple inflammation-related mediators to ACTH in patients with West syndrome has not previously been reported. This study aimed to explore the mechanisms of ACTH action by evaluating various cytokine responses to ACTH in patients with West syndrome.…”

mentioning

confidence: 96%

Serial Analysis of Multiple Serum Cytokine Responses to Adrenocorticotropic Hormone Therapy in Patients With West Syndrome

et al. 2018

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Adrenocorticotropic hormone (ACTH) therapy is effective for West syndrome; however, the underlying mechanism of action remains unknown. This study explored this mechanism in 5 Japanese patients with West syndrome, injected with ACTH for 28 days. Serum samples were obtained before and 30, 120, and 720 minutes after ACTH injection divided into an "early" (1-4 days) and a "late" (10-28 days) group. Responses to ACTH over time were analyzed by measuring the levels of 27 cytokines. In the early group, serum levels of interleukins-5, -9, and -17, basic fibroblast growth factor, interferon (IFN-γ), IFN-γ-inducible protein 10, chemokine ligand (CCL) 3 and 4, and platelet-derived growth factor were higher in all patients before ACTH administration than in the 720-minute time point. In the late group, no definite trend was observed except for decreased CCL2 levels after ACTH administration. These changes may correlate with mechanisms underlying the anticonvulsant effects of ACTH.

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Comparison of the serum cytokine levels before and after adrenocorticotropic hormone (ACTH) therapy in patients with infantile spasm

Abstract: Our study supports the recent idea that IL-6 and IL-17A are cytokines involved in the pathogenesis of infantile spasm.

Cited by 12 publications

References 20 publications

Spontaneous remission of West syndrome following a human herpesvirus 7 infection in a Chinese infant

Spontaneous remission of West syndrome following a human herpesvirus 7 infection in a Chinese infant

ACTH and PMX53 recover synaptic transcriptome alterations in a rat model of infantile spasms

Serial Analysis of Multiple Serum Cytokine Responses to Adrenocorticotropic Hormone Therapy in Patients With West Syndrome

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