Epilepsy, Tuberous Sclerosis Complex
Conditions
Keywords
epilepsy, tuberous sclerosis complex, resective epilepsy surgery
Brief summary
A prospective cohort studies to identify clinical seizure control, cognitive changes, and safety in resective epilepsy surgery in patients with TSC-related drug-resistant epilepsy.
Detailed description
Two hundred candidates are recruited and assigned to the control and surgery groups with 100 cases in each group, respectively. All patients will accept first-stage pre-enrollment evaluations and patients in surgery group will accept second-stage invasive evaluation. All patients will be advised to visit the hospitals for examination each year after enrollment. Seizure outcomes will be assessed with reference to patients' seizure diaries and caregivers' oral statements by both neurosurgeons and neurologists. All adverse effects will be classified according to the Common Terminology Criteria for Adverse Events (CTCAE), version 5.0 (2017). Statistical analyses were conducted using SPSS (version 26.0, IBM, USA), Stata (version 16.0, Stata Corp LLC, USA), and R (version 4.3.3, R Foundation for Statistical Computing, Austria). Outcomes were presented according to data type as percentages, mean ± standard deviation, or median (interquartile range, IQR). Multiple imputation was employed to address missing data for IQ and QOL at enrollment and the 2-year follow-up. A sensitivity analysis was conducted to assess the robustness of the multiple imputation results. The mi impute regress statement in Stata facilitated the multiple imputations. 20 T-tests were utilized for comparing continuous variables, with results reported as mean ± standard deviation. The Mann-Whitney U test was applied for non-normally distributed continuous variables, presenting results as median values and IQRs. Chi-square and Fisher's exact tests were used for univariate analyses. A significance level of p \< 0.05 was established. To account for differences in covariates between the surgery and medicine groups, propensity score matching (PSM) was performed. Based on PSM, Kaplan-Meier curves and the log-rank test were utilized to analyze time-to-seizure recurrence differences between the surgery and medicine groups. Additionally, multivariable Cox proportional hazards regression was applied to predict risk factors for postoperative seizure recurrence within a 51-month follow-up period after PSM.
Interventions
PROCEDURESurgery
Resective surgery included lobectomy (partial or total brain lobe resection), tuberectomy (epileptogenic tuber resection), and tuberectomy plus (resection of epileptogenic tuber and perituberal gyri). Tuberectomy was typically performed on the epileptogenic tuber within or near an eloquent area. Lobectomy was performed in patients with large epileptogenic tubers in the brain lobes. Multiple lobectomies, tuberectomies, tuberectomies plus, and lobectomy combined with tuberectomy/tuberectomies plus were further performed in patients with multiple epileptogenic tubers.
Sponsors
Shenzhen Children's Hospital
Tiantan Hospital, Capital Medical University
Peking University People's Hospital
Guangdong 999 Brain Hospital
Xinqiao Hospital, Amry Medical University
Shandong Provincial Hospital
West China Hospital
Henan Provincial People's Hospital
Shanghai Children's Hospital
First Medical Center, PLA General Hospital
Fourth Medical Center, PLA General Hospital
The First Affiliated Hospital of University of Science and Technology of China
Ruijin Hospital
First Affiliated Hospital Xi'an Jiaotong University
The Second Hospital of Hebei Medical University
The First Affiliated Hospital of Shanxi Medical University
Xuanwu Hospital, Beijing
The First Hospital of Jilin University
Xiangya Hospital of Central South University
Beijing Sanbo Brain Hospital
Nanjing Medical University
Beijing Children's Hospital
Study design
Observational model
COHORT
Time perspective
PROSPECTIVE
Eligibility
Sex/Gender
ALL
Age
2 Years to No maximum
Healthy volunteers
No
Inclusion criteria
* 2 years old and above, no gender restriction, TSC gene monitoring with or without abnormality * Diagnosis of tuberous sclerosis- related drug-resistant epilepsy * Epilepsy course for more than 1 year * Patients who have taken 3 or more reasonable choices with appropriate and tolerable antiepileptic drugs (excluding mTOR inhibitors and traditional Chinese medicine and prescriptions) had seizures more than 12 times in the 3 months before enrollment * The family members agreed to enroll and signed the informed consent.
Exclusion criteria
： * Obvious renal angiomyolipoma, pulmonary lymphoma leiomyomatosis, and subventricular giant cell astrocytoma * Abnormal heart, lung, liver, and kidney functions and coagulation function * Preoperative evaluation, it is considered that no surgical treatment is needed * The patient received other craniocerebral surgical treatment within 1 year during the follow-up period
Design outcomes
Primary
| Measure | Time frame | Description |
|---|---|---|
| % of patients with ILAE grading | 3 years | The grading made by ILAE for the classification of outcome with respect to epileptic seizures following epilepsy surgery. It contains a total of 6 levels. The higher the level, the worse the result. We will count the percentage of patients at each level. |
Secondary
| Measure | Time frame | Description |
|---|---|---|
| IQ | 3 years | Wechsler Preschool and Primary Scale of Intelligence（WPPSI-III），Wechsler Intelligence Scale for Children (WISC-IV) and Wechsler Adult Intelligence Scale (WAIS-IV)，3 scales will be used for different ages to measure IQ. Higher scores mean better outcomes. |
| Quality of Life: QOLIE-31 | 3 years | QOLIE-31 is suitable for patients 14 years and older. QOLCE-76 is applied to patients aged 2-13 by parents. |
Countries
China
Outcome results
None listed