Evidence map›Paper›PMID 37368068›Full record

ArticleChild's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery2023

Lhermitte-Duclos disease with concomitant KCNT2 gene mutation: report of an extremely rare combination.

Jeries Assi, Marianna Chyta, Ioannis Mavridis

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Article in Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

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1citing papers in PubMed
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1 · What the graph read from it

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1 citing paper in PubMed.

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4 · The record

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5 · Who and what money

Authors and funding

3 authors.

Jeries AssiSchool of Medicine, Faculty of Health Sciences, Democritus University of Thrace, Alexandroupolis, Greece.
Marianna ChytaSchool of Medicine, Faculty of Health Sciences, Democritus University of Thrace, Alexandroupolis, Greece.
Ioannis MavridisSchool of Medicine, Faculty of Health Sciences, Democritus University of Thrace, Alexandroupolis, Greece. inmavridis@gmail.com.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Lhermitte-Duclos disease (LDD) refers to cerebellar dysplastic gangliocytoma, a slow-growing tumor. Pathogenic variants of voltage-gated potassium channels have been associated with epilepsy of variable severity. These include the sodium-activated potassium channel subfamily T member 2 (KCNT2) gene, which encodes for pore-forming alpha subunits. KCNT2 gene mutations have been recently described to cause developmental and epileptic encephalopathies (DEEs). The purpose of the present article is to describe an extremely rare case of a young child who has both LDD and KCNT2 mutation. Our patient is an 11-year-old boy who presented with an absence episode, and his investigations revealed electroencephalography (EEG) abnormalities, LDD, and a heterozygous KCNT2 mutation. Regarding LDD patients, epileptic seizures have been reported in very few cases. Reports of patients with mutated KCNT2 variants are also extremely rare. It is for sure that LDD and KCNT2 mutation is an extremely rare combination. Although further follow-up is mandatory in order to draw safe conclusions for our case, the available data support that our patient is either the first reported case of a subclinical KCNT2 mutation or the first case of its clinical expression in late childhood so far.

Indexed as

Cerebellar NeoplasmsEpilepsyHamartoma Syndrome, MultipleChildHumansMagnetic Resonance ImagingMaleMutationPotassium Channels, Sodium-ActivatedSodiumKCNT2 protein, humanPotassium Channels, Sodium-ActivatedSodiumCerebellar dysplastic gangliocytomaDevelopmental and epileptic encephalopathyEpilepsyLhermitte-Duclos diseaseSodium-activated potassium channel subfamily T member 2

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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.