Efficacy of adjunctive vagus nerve stimulation in patients with Dravet syndrome: A meta-analysis of 68 patients

Dibué-Adjei, Maxine; Fischer, Igor; Steiger, Hans‐Jakob; Kamp, Marcel A.

doi:10.1016/j.seizure.2017.06.007

Cited by 44 publications

(29 citation statements)

References 42 publications

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“…Response to these treatment modalities has been mixed, but a recent meta-analysis of 13 case studies published in the literature reported that 53% of 68 patients with Dravet syndrome experienced at least 50% seizure reduction. 25 The exact time frame as to when to introduce such invasive treatment options is unclear but probably before the onset of severe brain dysfunction, which FDG-PET could demonstrate. In future studies, it would be also interesting to demonstrate if palliative treatment to reduce seizures could halt the progression of glucose hypometabolism.…”

Section: Discussionmentioning

confidence: 99%

Evolution of Brain Glucose Metabolic Abnormalities in Children With Epilepsy and SCN1A Gene Variants

et al. 2018

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Three children with drug-refractory epilepsy, normal magnetic resonance image (MRI), and a heterozygous SCN1A variant underwent 2-deoxy-2-[F]fluoro-d-glucose positron emission tomography (FDG-PET) scanning between age 6 months and 1 year and then at age 3 years 6 months to 5 years 5 months. Regional FDG uptake values were compared to those measured in age- and gender-matched pseudo-controls. At baseline, the brain glucose metabolic pattern in the SCN1A group was similar to that of the pseudo-controls. At follow-up, robust decreases of normalized FDG uptake was found in bilateral frontal, parietal and temporal cortex, with milder decreases in occipital cortex. Children with epilepsy and an SCN1A variant have a normal pattern of cerebral glucose metabolism at around 1 year of age but develop bilateral cortical glucose hypometabolism by age 4 years, with maximal decreases in frontal, parietal, and temporal cortex. This metabolic pattern may be characteristic of epilepsy associated with SCN1A variants and may serve as a biomarker to monitor disease progression and response to treatments.

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Section: Discussionmentioning

confidence: 99%

Evolution of Brain Glucose Metabolic Abnormalities in Children With Epilepsy and SCN1A Gene Variants

et al. 2018

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“…In a meta-analysis including 68 patients with Dravet syndrome treated with adjunctive VNS, 53% of patients experienced ≥ 50% reduction in seizures. 22 VNS was associated with a mean seizure reduction rate of 31% at 12 months in 8 pediatric patients with Dravet syndrome. 67…”

Section: Dravet Syndromementioning

confidence: 94%

The vagus afferent network: emerging role in translational connectomics

Hachem

Wong

Ibrahim

2018

Neurosurgical Focus

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Vagus nerve stimulation (VNS) is increasingly considered for the treatment of intractable epilepsy and holds potential for the management of a variety of neuropsychiatric conditions. The emergence of the field of connectomics and the introduction of large-scale modeling of neural networks has helped elucidate the underlying neurobiology of VNS, which may be variably expressed in patient populations and related to responsiveness to stimulation. In this report, the authors outline current data on the underlying neural circuitry believed to be implicated in VNS responsiveness in what the authors term the “vagus afferent network.” The emerging role of biomarkers to predict treatment effect is further discussed and important avenues for future work are highlighted.

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“…In DS children, there are many studies that display a efficacy of ≥ 50 % seizure reduction with a VNS implant [58][59][60]. A meta-analysis on the efficacy of VNS on 68 DS patients (13 studies) was reported by Dibue-Adjei et al [59] and showed an average of 55.2 % seizure reduction in these patients.…”

Section: Treatmentmentioning

confidence: 99%

A systematic review of adults with Dravet syndrome

Selvarajah

Ali

Marques

et al. 2021

Seizure

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Dravet Syndrome (DS) is a rare and severe infantile-onset epileptic encephalopathy. DS research focuses mainly on children. We did a systematic review, completed on January 18 th , 2021, examining the number of clinical DS studies. We show that there are 208 studies on children exclusively, 28 studies on adults exclusively, and 116 studies involving adults and children combined. This 7:1 ratio of children to adult studies exclusively shows the dearth of research that addresses long-term natural history of DS into adulthood.Through this systematic review, we examine the most up-to-date information in DS adults as it pertains to seizures, electroencephalogram, imaging, treatment, motor abnormalities, cognitive and social behavior outcomes, cardiac abnormalities, sleep disturbances, diagnosis in adults, and mortality.Overall, the frequency of seizures increases in the first decade of life and then myoclonic, atypical absences and focal seizures with impaired awareness tend to decrease in frequency or even disappear in adulthood. Adults tend to have a notable reduction in status epilepticus, especially after 30 years of age. Parkinsonian features were seen in patients as young as 19 years old and are more severe in older patients, suggesting a progression of the parkinsonian symptoms. In adulthood, patients continue to present with behavior problems, associated with a lower health-related quality of life. The leading reported cause of death in DS adults is Sudden Unexpected Death in Epilepsy (SUDEP).Further studies in older adults are needed to understand the long-term outcomes of patients with DS.

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Efficacy of adjunctive vagus nerve stimulation in patients with Dravet syndrome: A meta-analysis of 68 patients

Abstract: Vagus nerve stimulation appears to reduce seizure frequency in patients with DS. Based on this preliminary analysis, controlled trials of VNS in this rare condition using patient-centric outcome measures are indicated.

Cited by 44 publications

References 42 publications

Evolution of Brain Glucose Metabolic Abnormalities in Children With Epilepsy and SCN1A Gene Variants

Evolution of Brain Glucose Metabolic Abnormalities in Children With Epilepsy and SCN1A Gene Variants

The vagus afferent network: emerging role in translational connectomics

A systematic review of adults with Dravet syndrome

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