Evidence map›Paper›PMID 40331861›Full record

ArticleJournal of neurophysiology2025

Interneurons exhibit attenuated ectopic action potential firing in a severe neurodevelopmental disorder.

Sophie F Hill, Sophie R Liebergall, Eric R Wengert, Ethan M Goldberg, Brian B Theyel

Abstract read
In one paragraph

Article in Journal of neurophysiology, 2025. 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

5 authors.

Sophie F HillDivision of Neurology, Department of Pediatrics, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, United States.
Sophie R LiebergallDepartment of Neuroscience, The University of Pennsylvania Perelman School of Medicine, Philadelphia, Pennsylvania, United States.ORCID 0000-0001-6744-4835
Eric R WengertDivision of Neurology, Department of Pediatrics, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, United States.
Ethan M GoldbergDivision of Neurology, Department of Pediatrics, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, United States.
Brian B TheyelDepartment of Neuroscience, Brown University, Providence, Rhode Island, United States.ORCID 0000-0003-2299-2322

Funding

The Intellectual and Developmental Disabilities Research Center (IDDRC) at CHOP/PennP50HD105354 · NICHD · CHILDREN'S HOSP OF PHILADELPHIA · PI ERIC D MARSH, ROBERT Thomas SCHULTZ · 2021 to 2026
$9.2M
Interneuron axonopathy underlies circuit dysfunction in a mouse model of Dravet syndromeR01NS110869 · NINDS · CHILDREN'S HOSP OF PHILADELPHIA · PI ETHAN M GOLDBERG · 2019 to 2026
$3.5M
Origin, Mechanism, and Behavioral Context of Persistent Firing in Cortical Parvalbumin-Positive InterneuronsK08NS118114 · NINDS · BROWN UNIVERSITY · PI THEYEL, BRIAN · 2020 to 2024
$979k
Dravet Syndrome Foundation (DSF) Dravet Syndrome Foundation Postdoctoral FellowshipDravet Syndrome Foundation (DSF) Dravet Syndrome Foundation Research GrantNICHD NIH HHS P50 HD105354NINDS NIH HHS K08 NS118114NINDS NIH HHS R01 NS110869
6 · The paper itself

Abstract

Dravet syndrome (DS) is a severe neurodevelopmental disorder associated with treatment-resistant epilepsy and features of autism spectrum disorder due to loss of the voltage-gated sodium channel subunit Nav1.1. Recent work suggests that a pathogenic mechanism of DS is impaired action potential propagation along axons of cerebral cortex parvalbumin-positive fast-spiking GABAergic interneurons (PVINs). Here, we investigated another aspect of axonal physiology: action potentials generated in the distal axon, known as "ectopic" action potentials (EAPs). We hypothesized that EAP frequency could be a proxy for the excitability of the distal axon and that EAPs would be attenuated in neocortical layer 2/3 PVINs from DS mice due to axonal dysfunction. We identified reduced EAP generation in DS PVINs at both

Indexed as

Action PotentialsEpilepsies, MyoclonicGABAergic NeuronsInterneuronsNeocortexAnimalsAxonsDisease Models, AnimalFemaleMaleMiceMice, Inbred C57BLNAV1.1 Voltage-Gated Sodium ChannelParvalbuminsNAV1.1 Voltage-Gated Sodium ChannelParvalbuminsaction potentialsDravet syndromeectopic action potentialsepilepsyretroaxonal barrage

Identifiers

PMID40331861
PMCPMC12151314

What OpenQuestion holds

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Registered trials

None linked

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.