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Efficacy of ketogenic diet in CDKL5-related epilepsy: a single arm meta-analysis
Jie Zhang1, Jiayi Ma1, Xuting Chang1
1Department of Pediatrics, Peking University First Hospital, No. 1 Xi'an Men Street, West District, Beijing, 100034, China.
Background:
Drug-resistant epilepsy is one of the most important features of cyclin-dependent kinase-like 5 (CDKL5) deficiency disorder. The ketogenic diet (KD) may be effective for patients with CDKL5-related epilepsy, but there is little high-quality evidence to confirm the efficacy. This meta-analysis investigated the efficacy and safety of KD in CDKL5-related epilepsy.
Methods:
The PubMed, Embase, Web of Science, Cochrane Library, WanFang, CNKI and VIP databases were searched for relevant studies published up to January 1, 2022. Two reviewers independently screened the literature according to inclusion and exclusion criteria and evaluated the bias risk of the included studies. Meta-analysis was performed using Review Manager 5.3 software.
Results:
A total of 12 retrospective studies involving 193 patients met the inclusion criteria. Meta-analysis revealed that the definite responder rate to KD in the treatment of CDKL5-related epilepsy was 18.0% [95% CI (0.07, 0.67)], with no statistical heterogeneity among studies (I2 = 0%, P = 0.45). The clinical responder rate was 50.5% [95% CI (0.75, 1.39)], and there was no statistical heterogeneity among all studies (I2 = 46%, P = 0.05). Subgroup analysis showed that there was no significant difference in the clinical responder rate between the two groups with seizure onset age before and after 1 month (P = 0.14). Only one study mentioned adverse reactions, and the incidence of adverse reactions was 78.3% (18/23). Constipation and vomiting were the main manifestations, implying a high incidence of gastrointestinal adverse reactions.
Conclusions:
The definite responder rate to KD in CDKL5-related epilepsy was 18%, and the gastrointestinal adverse reactions were probably common in these patients. All the studies included in the meta-analysis were retrospective, and most of them had small sample sizes. Additional high-quality studies are needed to confirm the efficacy and tolerance of KD in CDKL5-related epilepsy.
Insights
The ketogenic diet (KD) shows a definite responder rate of 18% for drug-resistant epilepsy in cyclin-dependent kinase-like 5 (CDKL5) deficiency disorder. Gastrointestinal side effects like constipation and vomiting are common, necessitating further high-quality research.
Area of Science:
- Neurology
- Genetics
- Metabolic Disorders
Background:
- Cyclin-dependent kinase-like 5 (CDKL5) deficiency disorder is characterized by drug-resistant epilepsy.
- The ketogenic diet (KD) is a potential therapeutic option for CDKL5-related epilepsy, but high-quality evidence is limited.
Approach:
- A meta-analysis was conducted on 12 retrospective studies involving 193 patients with CDKL5 deficiency disorder.
- Literature search was performed across major databases up to January 2022.
- Data were analyzed to assess the efficacy and safety of KD.
Key Points:
- The definite responder rate to KD was 18.0% (95% CI: 0.07, 0.67) with no statistical heterogeneity.
- The clinical responder rate was 50.5% (95% CI: 0.75, 1.39) with no significant heterogeneity.
- Adverse reactions, primarily gastrointestinal (constipation, vomiting), occurred in 78.3% of patients in one study.
Conclusions:
- KD demonstrates an 18% definite responder rate for CDKL5-related epilepsy, with frequent gastrointestinal adverse reactions.
- Current evidence is based on retrospective studies with small sample sizes.
- High-quality research is required to validate KD's efficacy and tolerability in CDKL5 deficiency disorder.
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