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Record W4412714313 · doi:10.1016/j.abb.2025.110569

Sacsin deletion decreases cell viscoelasticity and motility in a glial cell model of autosomal recessive spastic ataxia of Charlevoix Saguenay

2025· article· en· W4412714313 on OpenAlexaboutno aff
Fernanda Murtinheira, João Belo, Ana Sofia Boasinha, Tiago T. Robalo, Vukosava Milic Torres, Francisco Pinto, Constança Pimenta, Patrícia Georgina Garcia do Nascimento, Mário S. Rodrigues, Federico Herrera

Bibliographic record

VenueArchives of Biochemistry and Biophysics · 2025
Typearticle
Languageen
FieldNeuroscience
TopicGenetic Neurodegenerative Diseases
Canadian institutionsnot available
FundersEuropean Regional Development FundFundação para a Ciência e a TecnologiaResearch Executive AgencyEuropean Research Executive AgencyRede Nacional de Espectrometria de MassaEuropean CommissionAtaxia UK
KeywordsMotilityAtaxiaViscoelasticityCellSpasticBiologyNeuroscienceGeneticsMedicinePhysicsCerebral palsyPhysical medicine and rehabilitation

Abstract

fetched live from OpenAlex

Autosomal recessive spastic ataxia of Charlevoix-Saguenay (ARSACS) is a movement disorder caused by loss-of-function mutations in the sacsin gene. The most common hallmark of this disease is the disruption of intermediate filament networks in cells as diverse as neurons, kidney cells, fibroblasts, astroglia and microglia. Intermediate filaments are the main filaments responsible for the mechanical and viscoelastic properties of cells and tissues, but these have never been investigated in the context of ARSACS. Here, we analyzed the consequences of sacsin loss on the mechanical functions of astroglial-like C6 cells. The phenotype of C6 Sacs−/− cells was analyzed by immunocytochemistry, electron microscopy, mass spectrometry, atomic force microscopy and motility/proliferation assays. C6 Sacs−/− cells presented an abnormal cytoskeletal and organelle distribution, global proteome alterations linked to cell motility and mechanics, a significant decrease in cell elasticity in the cytoplasm, and a striking reduction in cell motility. These mechanical alterations in glial-like cells could be especially relevant for neuroinflammation and glial scar formation upon CNS injury. Our results support a possible role for alterations in glial functions in ARSACS and provide new tools for understanding the glial-specific mechanisms involved in this movement disorder. • Glial-like cells lacking sacsin show proteome-wide mechanical alterations. • Sacsin deletion causes cytoplasmic disorganization of Golgi apparatus and plectin. • Sacsin deletion decreases viscoelasticity in cytoplasmic but not nuclear regions. • Sacsin deletion decreases the motility of cells in culture.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.998
Threshold uncertainty score0.005

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0010.001
Insufficient payload (model declined to judge)0.0010.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.010
GPT teacher head0.228
Teacher spread0.218 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designBench or experimental
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations0
Published2025
Admission routes1
Has abstractyes

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