Evidence map›Paper›PMID 41361856›Full record

ArticleActa neuropathologica communications2025

The profiling of extracellular vesicle subtypes in Huntington's disease brains identifies Alix as a novel marker of neuropathology.

Rocío Pérez-González, Anna Vázquez-Oliver, Nil Salvat-Rovira, Saül Martínez-Horta, Elisa Rivas-Asensio, Eva Borràs, Samanta Ortuño-Miquel, María Sánchez-Carcelén, Marta Garcia-Forn, Frederic Sampedro and 4 more

Abstract read
In one paragraph

Article in Acta neuropathologica communications, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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0cells of the map it votes in
0citing papers in PubMed
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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

0 citing papers in PubMed.

No citing paper in PubMed yet.

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

14 authors.

Rocío Pérez-GonzálezInstituto de Investigación Sanitaria y Biomédica de Alicante (ISABIAL), Hospital Universitario Doctor Balmis de Alicante, Av. Pintor Baeza 12, 03010, Alicante, Spain. perez_roc@isabial.es.
Anna Vázquez-OliverCentro de Investigación en Red-Enfermedades Neurodegenerativas (CIBERNED), 28031, Madrid, Spain.
Nil Salvat-RoviraCentro de Investigación en Red-Enfermedades Neurodegenerativas (CIBERNED), 28031, Madrid, Spain.
Saül Martínez-HortaCentro de Investigación en Red-Enfermedades Neurodegenerativas (CIBERNED), 28031, Madrid, Spain.
Elisa Rivas-AsensioCentro de Investigación en Red-Enfermedades Neurodegenerativas (CIBERNED), 28031, Madrid, Spain.
Eva BorràsCenter for Genomic Regulation (CRG), Barcelona Institute of Science and Technology, 08003, Barcelona, Spain.
Samanta Ortuño-MiquelInstituto de Investigación Sanitaria y Biomédica de Alicante (ISABIAL), Hospital Universitario Doctor Balmis de Alicante, Av. Pintor Baeza 12, 03010, Alicante, Spain.
María Sánchez-CarcelénInstituto de Investigación Sanitaria y Biomédica de Alicante (ISABIAL), Hospital Universitario Doctor Balmis de Alicante, Av. Pintor Baeza 12, 03010, Alicante, Spain.
Marta Garcia-FornCentro de Investigación en Red-Enfermedades Neurodegenerativas (CIBERNED), 28031, Madrid, Spain.
Frederic SampedroNeuroradiology Section, Radiology Department, Hospital Vall d'Hebron - Institut de Diagnostic Per la Imatge, 08035, Barcelona, Spain.
Jesús Pérez-PérezCentro de Investigación en Red-Enfermedades Neurodegenerativas (CIBERNED), 28031, Madrid, Spain.
Eduard SabidóCenter for Genomic Regulation (CRG), Barcelona Institute of Science and Technology, 08003, Barcelona, Spain.
Esther Pérez-NavarroCentro de Investigación en Red-Enfermedades Neurodegenerativas (CIBERNED), 28031, Madrid, Spain.
Jaime KulisevskyCentro de Investigación en Red-Enfermedades Neurodegenerativas (CIBERNED), 28031, Madrid, Spain.

Funding

Agència de Gestió d'Ajuts Universitaris i de Recerca 2021-SGR2021-01225Centro de Investigación Biomédica en Red sobre Enfermedades Neurodegenerativas CB06/05/0047European Huntington's Disease Network Seed fund project 1119Instituto de Salud Carlos III CP21/00009Ministerio de Ciencia e Innovación MCIN/AEI/10.13039/501100011033Ministerio de Ciencia e Innovación PID2019-106447RB-100
6 · The paper itself

Abstract

backgroundHuntington's disease (HD) is the most frequent autosomal dominant neurodegenerative disorder, which is caused by a CAG repeat expansion in the HTT gene. Despite its well-defined genetic origin, there is currently no cure, and reliable biomarkers for disease progression and pathophysiology remain limited. Mutant huntingtin protein accumulates in endosomal compartments, disrupting endosomal trafficking and potentially affecting the biogenesis, release, and cargo of exosomes-extracellular vesicles (EVs) derived from the endosomal pathway. However, the role of exosomes in HD pathogenesis and their potential as biomarkers has been underexplored. In this work, we investigated whether the levels and content of small EV subpopulations, including exosomes, are altered in the brains of HD patients.

methodsWe analyzed two distinct subpopulations of small EVs from the striatum and cortex of postmortem HD brains at early and advanced neuropathological stages, as well as from age-matched controls. EVs were isolated by differential ultracentrifugation and high-resolution iodixanol density gradient centrifugation, and analyzed by Western blotting, electron microscopy, NTA, and proteomics using mass spectrometry. EV secretion was also analyzed in primary fibroblasts derived from HD patients and healthy controls.

resultsMass spectrometry data revealed HD-associated alterations in EV protein content, particularly proteins related to the endosomal system. Our data also indicate that the level of ectosomes increased in the HD cortex, whereas exosomes were reduced in the HD striatum compared to controls. In terms of EV content, EVs from HD brains showed increased levels of Annexin A2 and decreased levels of Alix, a key component of the endosomal sorting complex required for transport (ESCRT). Alix depletion in EVs mirrored a progressive reduction of Alix in brain tissue, correlating with disease severity based on Vonsattel staging. In vitro, HD fibroblasts secreted EVs with reduced Alix content, despite no significant difference in cellular Alix levels compared to controls.

conclusionsThese findings highlight disease-specific changes in EV populations and cargo in HD, and identify Alix as a potential neuropathological marker. This study advances our understanding of the role of brain-derived EVs in HD and underscores their potential utility in biomarker discovery.

Indexed as

BrainCalcium-Binding ProteinsCell Cycle ProteinsEndosomal Sorting Complexes Required for TransportExtracellular VesiclesHuntington DiseaseAdultAgedBiomarkersExosomesFemaleHumansMaleMiddle AgedBiomarkersCalcium-Binding ProteinsCell Cycle ProteinsEndosomal Sorting Complexes Required for TransportPDCD6IP protein, humanAlixAnnexin A2EctosomesExosomesHuntington’s diseaseNeurodegeneration

Identifiers

PMID41361856
PMCPMC12896033

What Socratic holds

Textmetadata
LicenceCC BY
Read underepoch 390

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 Socratic graph.