Evidence map›Paper›PMID 41384922›Full record

ReviewJournal of Huntington's disease2026

Insights into neurodevelopmental features of Huntington's disease from stem cell-derived models including organoids.

Mariana Sierra, Rachael Powers, Nikolas Grotewold, Henry Paulson

Abstract readReview
In one paragraph

Review in Journal of Huntington's disease, 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. Review
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

4 authors.

Mariana SierraDepartment of Neurology, University of Michigan, Ann Arbor, MI, USA.
Rachael PowersDepartment of Neurology, University of Michigan, Ann Arbor, MI, USA.ORCID 0000-0001-6425-1341
Nikolas GrotewoldDepartment of Neurology, University of Michigan, Ann Arbor, MI, USA.ORCID 0000-0003-2437-6451
Henry PaulsonDepartment of Neurology, University of Michigan, Ann Arbor, MI, USA.ORCID 0000-0002-0382-7535

Funding

MICHIGAN MEDICAL SCIENTIST TRAINING PROGRAMT32GM007863 · NIGMS · UNIVERSITY OF MICHIGAN AT ANN ARBOR · PI COLLINS, KATHLEEN L. · 1985 to 2024
$38.3M
CELLULAR AND MOLECULAR BIOLOGY AT MICHIGANT32GM007315 · NIGMS · UNIVERSITY OF MICHIGAN AT ANN ARBOR · PI PUTHENVEEDU, MANOJKUMAR A · 1985 to 2021
$12.8M
Mechanisms of neurodegenerative diseases: intersections with ubiquitin pathwaysR35NS122302 · NINDS · UNIVERSITY OF MICHIGAN AT ANN ARBOR · PI Henry L Paulson · 2021 to 2026
$6.4M
NIGMS NIH HHS T32 GM007315NIGMS NIH HHS T32 GM007863NINDS NIH HHS R35 NS122302
6 · The paper itself

Abstract

Two- and three-dimensional (2D and 3D) cell models derived from human stem cells have shed light on a wide range of molecular and cellular features of Huntington's disease (HD). Here we review the use of human stem cell-derived models to explore neurodevelopmental contributions to HD. We provide a timeline of key advances made in 2D and 3D model systems, ranging from differentiated monocultures to brain-like organoids and assembloids. Models along this spectrum have advanced our understanding of various disease-associated characteristics including disease protein (huntingtin) aggregation, somatic repeat instability, transcriptional dysregulation, perturbations in neurodevelopmental staging, and neural circuitry. We highlight recent findings in brain-like organoids which, despite being a relatively recent innovation, are proving to be a promising tool with which to study aberrant neurodevelopmental features of HD. All models have their limitations, and we compare and contrast the utility and limitations of various stem cell-based methods to study HD. Finally, we speculate on future advances employing advanced computational and transcriptomic methods that will expand the power of 3D model systems for the study of HD and related neurodegenerative disorders.

Indexed as

BrainHuntington DiseaseOrganoidsStem CellsAnimalsHumansNeurodevelopmentneurodegenerationneurodevelopmentorganoidspolyglutamine diseasesrepeat expansion diseases

Identifiers

PMID41384922
PMCPMC12910477

What OpenQuestion holds

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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 OpenQuestion graph.