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Record W3197268513 · doi:10.1136/jnnp-2021-ehdn.123

I09 In vivo mtHTT protein reduction in the CNS and periphery by passive immunization with the monoclonal antibody C6–17

2021· article· en· W3197268513 on OpenAlexaff
Stefan Bartl, Yuanyun Xie, Nalini Potluri, Benjamin Gordon, Alicia Willenberg, Katja Balash, Abid Oueslati, Michela Parth, Nina Salhat, Alberto Siddu, Oskar W. Smrzka, Francesca Cicchetti, Günther Straffler, Michael R. Hayden, Amber L. Southwell

Bibliographic record

Venuenot available
Typearticle
Languageen
FieldNeuroscience
TopicGenetic Neurodegenerative Diseases
Canadian institutionsCentre hospitalier de l'Université LavalUniversity of British Columbia
Fundersnot available
KeywordsMonoclonal antibodyHuntingtinBiologyIn vivoHuntingtin ProteinAntibodyGene knockinCell biologyImmunologyPathologyMolecular biologyMutantMedicineGeneticsGene

Abstract

fetched live from OpenAlex

Huntington’s disease (HD) is a hereditary neurodegenerative disorder characterized by changes in personality, cognition and motor control. The cardinal neuropathological hallmark of this disease is the massive atrophy of the striatum resulting from neuronal dysfunction and loss, but which extends to other areas of the brain as well as peripheral organs. The genetic mutation underlying HD originates in Exon1 of the huntingtin gene gives rise to a toxic/mutated form of the huntingtin protein (mtHTT). The mtHTT protein is ubiquitously expressed but also exhibits the ability to propagate from cell-to-cell to disseminate pathology, a property which may serve as a new therapeutic focus. We have developed a monoclonal antibody C6-17 targeting a particularly exposed region close to the aa586 Caspase 6 cleavage site of the huntingtin protein and, as recently published, mAB C6-17 is able to block cell-to-cell propagation of mutated HTT in vitro. In order to reduce the burden of the mutant protein in vivo, we queried whether the freely accessible and extracellular mtHTT can be targeted by an antibody. In POC experiments, using the transgenic animal model YAC128, we found that after 3 months mAB C6-17 treatment the circulating mtHTT in the peripheral as well as in the CNS tissues was reduced. Further, we could demonstrate the presence of active mAB C6-17 in PBS/heparin perfused peripheral and CNS tissues. The mAB C6-17 treated YAC128 animals showed benefits in body wight and motor behaviors and we could observe a delay in the HD disease progression. The obtained in vivo results provide the first POC data for the feasibility and efficacy of an antibody-based anti-mtHTT approach and suggest this therapeutic strategy as a potential new HD treatment possibility.

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 score0.001
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)0.0010.001

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 head0.012
GPT teacher head0.248
Teacher spread0.236 · 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

Citations1
Published2021
Admission routes1
Has abstractyes

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