Evidence map›Paper›PMID 38638299›Full record

ArticleFrontiers in cellular neuroscience2024

Abnormal cell sorting and altered early neurogenesis in a human cortical organoid model of Protocadherin-19 clustering epilepsy.

Wei Niu, Lu Deng, Sandra P Mojica-Perez, Andrew M Tidball, Roksolana Sudyk, Kyle Stokes, Jack M Parent

Open access · goldAbstract read
In one paragraph

Article in Frontiers in cellular neuroscience, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 8 papers.

0numbers the graph read from it
0cells of the map it votes in
8citing papers in PubMed
4.4field-weighted citation impact, top 6% of its field
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

8 citing papers in PubMed, 8 citations in OpenAlex.

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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

7 authors at 2 institutions in 2 countries.

Wei NiuDepartment of Neurology, University of Michigan, Ann Arbor, MI, United States.
Lu DengDepartment of Neurology, University of Michigan, Ann Arbor, MI, United States.
Sandra P Mojica-PerezDepartment of Neurology, University of Michigan, Ann Arbor, MI, United States.
Andrew M TidballDepartment of Neurology, University of Michigan, Ann Arbor, MI, United States.
Roksolana SudykDepartment of Neurology, University of Michigan, Ann Arbor, MI, United States.
Kyle StokesDepartment of Neurology, University of Michigan, Ann Arbor, MI, United States.
Jack M ParentDepartment of Neurology, University of Michigan, Ann Arbor, MI, United States.
Michigan United · USUniversity of Michigan–Ann Arbor · US

Funding

Proteins to Cell SystemsU54NS117170 · NINDS · UNIVERSITY OF MICHIGAN AT ANN ARBOR · PI ISOM, LORI L., PARENT, JACK M · 2020 to 2024
$11.9M
NINDS NIH HHS U54 NS117170
6 · The paper itself

Abstract

Introduction: Protocadherin-19 ( Methods: To overcome these challenges and model mosaic Results: We found that PCDH19 is highly expressed in early (days 20-35) WT neural rosettes where it co-localizes with N-Cadherin in ventricular zone (VZ)-like regions. Mosaic PCE hCOs displayed abnormal cell sorting in the VZ with KO and WT cells completely segregated. This segregation remained robust when WT:KO cells were mixed at 2:1 or 1:2 ratios. PCE hCOs also exhibited altered expression of PCDH19 (in WT cells) and N-Cadherin, and abnormal deep layer neurogenesis. None of these abnormalities were observed in hCOs generated by mixing only WT or only KO (modeling male carrier) cells. Discussion: Our results using the mosaic PCE hCO model suggest that PCDH19 plays a critical role in human VZ radial glial organization and early cortical development. This model should offer a key platform for exploring mechanisms underlying PCE-related cortical hyperexcitability and testing of potential precision therapies.

Indexed as

brain organoidCRISPR/CAS9 genome editingmosaicismneurodevelopmentPCDH19-clustering epilepsyProtocadherin-19random X-chromosome inactivation

Identifiers

PMID38638299
PMCPMC11024992
OpenAlexW4393935509

What Socratic holds

Textmetadata
LicenceCC BY
Read underepoch 390

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 Socratic graph.