Evidence map›Paper›PMID 42360057›Full record

SynthesisEpilepsia2026

SUDEP and mortality in developmental and epileptic encephalopathies: A meta-analysis of randomized clinical trials and extension studies.

Pierludovico Moro, Maria Sole Borioni, Adolfo Mazzeo, Enrico Cocchi, Carlo Di Bonaventura, Emanuele Cerulli Irelli

Abstract readMeta-Analysis
In one paragraph

Synthesis in Epilepsia, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

6 authors.

Pierludovico MoroDepartment of Human Neurosciences, Sapienza University, Rome, Italy.ORCID https://orcid.org/0009-0007-7683-5310
Maria Sole BorioniDepartment of Human Neurosciences, Sapienza University, Rome, Italy.ORCID https://orcid.org/0009-0002-5450-483X
Adolfo MazzeoDepartment of Human Neurosciences, Sapienza University, Rome, Italy.ORCID https://orcid.org/0000-0002-7014-6617
Enrico CocchiDepartment of Medical and Surgical Sciences, University of Bologna, Bologna, Italy.ORCID https://orcid.org/0000-0002-7532-956X
Carlo Di BonaventuraDepartment of Human Neurosciences, Sapienza University, Rome, Italy.ORCID https://orcid.org/0000-0003-1890-5409
Emanuele Cerulli IrelliDepartment of Human Neurosciences, Sapienza University, Rome, Italy.ORCID https://orcid.org/0000-0003-3906-555X

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

objectiveDevelopmental and epileptic encephalopathies (DEEs) are associated with high premature mortality and increased risk of sudden unexpected death in epilepsy (SUDEP). However, epidemiological data remain limited, particularly for specific syndromes such as Dravet syndrome (DS), Lennox-Gastaut syndrome (LGS), and infantile epileptic spasms syndrome (IESS). To address this gap, we conducted a Bayesian meta-analysis of randomized controlled trials (RCTs) and corresponding long-term open-label extension (OLE) studies.

methodsAll-cause mortality and SUDEP incidence rates were estimated using hierarchical Bayesian models with moderately informative priors derived from the literature. A negative binomial likelihood was used for all-cause mortality, whereas a Poisson likelihood was used for SUDEP. Subgroup analysis were conducted for DS, LGS, and IESS.

resultsThirty-seven RCTs and 15 OLE studies were included, comprising 3757 patients with DEEs (2221 LGS, 998 DS, 369 IESS, and 169 other DEEs). Across pooled data, 37 deaths occurred, including 2 definite, 12 probable, and 1 possible SUDEP. In the overall DEE population, the Bayesian posterior median estimated mortality rate was 8.76 per 1000 person-years (PY; 95% credible interval [CrI], 5.50-13.89), and SUDEP rate was 4.32 per 1000 PY (95% CrI, 2.66-6.64). In LGS, mortality was 7.05 per 1000 PY and SUDEP was 3.4 per 1000 PY. In DS, mortality was 8.0 per 1000 PY, with a SUDEP rate of 7.59 per 1000 PY. In IESS, mortality was 9.41 per 1000 PY and SUDEP was 2.79 per 1000 PY. SIGNIFICANCE: SUDEP accounted for approximately half of deaths in the overall DEE population and in LGS, with a proportionally greater contribution in DS. Potential trial-related underestimation should be considered when interpreting these findings, as features of trial-based datasets could have contributed to lower observed mortality and SUDEP estimates. Prospective population-based studies are needed to more accurately define the real-world incidence of SUDEP in DEEs.

Indexed as

Epilepsies, MyoclonicEpilepsyLennox Gastaut SyndromeSpasms, InfantileSudden Unexpected Death in EpilepsyBayes TheoremHumansIncidenceRandomized Controlled Trials as TopicchildhoodDravet syndromeepilepsyinfantile epileptic spasm syndromeLennox–Gastaut syndrome

Identifiers

PMID42360057
PMCPMC13592499

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.