ABSTRACT
BACKGROUND: Drug-resistant epilepsy (DRE) is a complex medical condition often requiring resective surgery and/or some form of neurostimulation. In recent years responsive neurostimulation (RNS) has shown promising results in adult DRE, however there is a paucity of information regarding outcomes of RNS among pediatric patients treated with DRE. In this individual patient data meta-analysis (IPDMA) we seek to elucidate the effects RNS has on the pediatric population. METHODS: Literature regarding management of pediatric DRE via RNS was reviewed in accordance with individual patient data meta-analysis guidelines. Four databases were searched with keywords ((Responsive neurostimulation) AND (epilepsy)) through December of 2022. From 1624 retrieved full text studies, 15 were ultimately included affording a pool of 98 individual participants. RESULTS: The median age at implantation was 14 years (n = 95) with 42% of patients having undergone prior resective epilepsy surgery, 18% with prior vagus nerve stimulation (VNS), and 1% with prior RNS. At a median follow up time 12 months, median percent seizure reduction was 75% with 57% of patients achieving Engel Class < 2 outcome, 9.7% of which achieved seizure freedom. We report a postoperative complication rate of 8.4%, half of which were device-related infections. Magnetic resonance imaging (MRI)-negative cases were negatively associated with magnitude of seizure reduction, and direct targeting techniques were associated with stronger treatment response when compared to other methods. CONCLUSIONS: This review suggests RNS to be an effective treatment modality for pediatric patients with a postoperative complication rate comparable to that of RNS in adults. Investigation of prognostic clinical variables should be undertaken to augment patient selection. Last, multi-institutional prospective study of long-term effects of RNS on pediatric patients would stand to benefit clinicians in the management of pediatric DRE.
Subject(s)
Deep Brain Stimulation , Drug Resistant Epilepsy , Epilepsy , Adult , Humans , Child , Adolescent , Drug Resistant Epilepsy/therapy , Deep Brain Stimulation/methods , Prospective Studies , Epilepsy/therapy , Treatment Outcome , Seizures/therapy , Postoperative Complications/therapyABSTRACT
OBJECTIVE: In recent years, the treatment of drug-resistant epilepsy (DRE) has made greater use of surgery and expanded options for neurostimulation or neuromodulation. Up to this point, responsive neurostimulation (RNS) has been very promising but has mainly used only the cortex as a target. In this individual patient data meta-analysis (IPDMA), the authors sought to establish if a novel RNS target, the thalamus, can be used to treat DRE. METHODS: The literature regarding the management of DRE by targeting the thalamus with RNS was reviewed per IPDMA guidelines. Five databases were searched with keywords [((Responsive neurostimulation) OR (RNS)) AND ((thalamus) OR (thalamic) OR (Deep-seated) OR (Diencephalon) OR (limbic))] in March 2022. RESULTS: The median (interquartile range) age at implantation was 17 (13.5-27.5) years (n = 42) with an epilepsy duration of 12.1 (5.8-15.3) years. In total, 52.4% of patients had previously undergone epilepsy surgery, 28.6% had prior vagus nerve stimulation, and 2.4% had prior RNS. The median preimplant seizure frequency was 12 per week. The median seizure reduction at last follow-up was 73%. No study in this IPDMA reported complications, although 7 cases (16.3%) did require reoperation. Behavioral improvements and reduced antiepileptic drug dose or quantity were reported for 80% and 28.6% of patients, respectively. CONCLUSIONS: This review indicates that thalamic RNS may be safe and effective for treating DRE. Long-term and controlled studies on thalamic RNS for DRE would further elucidate this technique's potential benefits and complications and help guide clinical judgment in the management of DRE.
Subject(s)
Deep Brain Stimulation , Drug Resistant Epilepsy , Epilepsy , Humans , Adolescent , Young Adult , Adult , Drug Resistant Epilepsy/therapy , Thalamus , Epilepsy/therapy , Seizures/therapyABSTRACT
BACKGROUND: Despite the well-documented utility of responsive neurostimulation (RNS, NeuroPace) in adult epilepsy patients, literature on the use of RNS in children is limited. OBJECTIVE: To determine the real-world efficacy and safety of RNS in pediatric epilepsy patients. METHODS: Patients with childhood-onset drug-resistant epilepsy treated with RNS were retrospectively identified at 5 pediatric centers. Reduction of disabling seizures and complications were evaluated for children (<18 yr) and young adults (>18 yr) and compared with prior literature pertaining to adult patients. RESULTS: Of 35 patients identified, 17 were <18 yr at the time of RNS implantation, including a 3-yr-old patient. Four patients (11%) had concurrent resection. Three complications, requiring additional surgical interventions, were noted in young adults (2 infections [6%] and 1 lead fracture [3%]). No complications were noted in children. Among the 32 patients with continued therapy, 2 (6%) achieved seizure freedom, 4 (13%) achieved ≥90% seizure reduction, 13 (41%) had ≥50% reduction, 8 (25%) had <50% reduction, and 5 (16%) experienced no improvement. The average follow-up duration was 1.7 yr (median 1.8 yr, range 0.3-4.8 yr). There was no statistically significant difference for seizure reduction and complications between children and young adults in our cohort or between our cohort and the adult literature. CONCLUSION: These preliminary data suggest that RNS is well tolerated and an effective off-label surgical treatment of drug-resistant epilepsy in carefully selected pediatric patients as young as 3 yr of age. Data regarding long-term efficacy and safety in children will be critical to optimize patient selection.
Subject(s)
Deep Brain Stimulation , Drug Resistant Epilepsy , Epilepsy , Child , Cohort Studies , Drug Resistant Epilepsy/surgery , Epilepsy/therapy , Humans , Retrospective Studies , Seizures/therapy , Young AdultSubject(s)
Abducens Nerve Diseases/diagnosis , Gram-Positive Bacterial Infections/diagnosis , Mastoiditis/diagnosis , Otitis Media/diagnosis , Pneumonia, Bacterial/diagnosis , Trigeminal Neuralgia/diagnosis , Actinomycosis/diagnosis , Ampicillin/therapeutic use , Anti-Bacterial Agents/therapeutic use , Child, Preschool , Coinfection , Diagnosis, Differential , Fatigue/etiology , Fusobacterium Infections/diagnosis , Fusobacterium nucleatum , Gram-Positive Bacterial Infections/drug therapy , Humans , Male , Peptostreptococcus , Pneumonia, Bacterial/drug therapy , Recurrence , Sulbactam/therapeutic use , Syndrome , Weight LossABSTRACT
Investigators from the Danish Epilepsy Center the frequency of SLC2A1 mutations in a cohort of patients with different types of epilepsies.
ABSTRACT
Investigators from Children's Hospital Aschaffenburg, Germany; University of Manchester, Manchester, United Kingdom; University Children's Hospital Tübingen, Tübingen, Germany; and other international centers studied prognostic factors in optic neuritis.
