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Record W3137362953 · doi:10.1186/s40478-021-01132-7

Two distinct conformers of PrPD type 1 of sporadic Creutzfeldt–Jakob disease with codon 129VV genotype faithfully propagate in vivo

2021· article· en· W3137362953 on OpenAlexaff
Ignazio Calì, Juan Carlos Espinosa, Satish Kumar Nemani, Alba Marín‐Moreno, Manuel V. Camacho, Rabail Aslam, Tetsuyuki Kitamoto, Brian S. Appleby, Juan María Torres, Pierluigi Gambetti

Bibliographic record

VenueActa Neuropathologica Communications · 2021
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicPrion Diseases and Protein Misfolding
Canadian institutionsUniversity of Alberta
FundersNational Institute of Neurological Disorders and StrokeNational Institute of Allergy and Infectious DiseasesAgencia Estatal de InvestigaciónBritton FundCenters for Disease Control and PreventionNational Institutes of HealthFundació la Marató de TV3National Institute on AgingInstituto Nacional de Investigación y Tecnología Agraria y Alimentaria
KeywordsGene isoformPhenotypeGenotypeBiologyValineMolecular biologyGenePrion proteinGenetically modified mouseIn vivoPRNPTransgeneGeneticsDiseasePathologyAmino acidMedicine

Abstract

fetched live from OpenAlex

Abstract Current classifications of sporadic Creutzfeldt–Jakob disease (sCJD) identify five subtypes associated with different disease phenotypes. Most of these histopathological phenotypes (histotypes) co-distribute with distinct pairings of methionine (M)/valine (V) genotypes at codon 129 of the prion protein (PrP) gene and the type (1 or 2) of the disease-associated PrP (PrP D ). Types 1 and 2 are defined by the molecular mass (~ 21 kDa and ~ 19 kDa, respectively) of the unglycosylated isoform of the proteinase K-resistant PrP D (resPrP D ). We recently reported that the sCJDVV1 subtype (129VV homozygosity paired with PrP D type 1, T1) shows an electrophoretic profile where the resPrP D unglycosylated isoform is characterized by either one of two single bands of ~ 20 kDa (T1 20 ) and ~ 21 kDa (T1 21 ), or a doublet of ~ 21–20 kDa (T1 21−20 ). We also showed that T1 20 and T1 21 in sCJDVV have different conformational features but are associated with indistinguishable histotypes. The presence of three distinct molecular profiles of T1 is unique and raises the issue as to whether T1 20 and T1 21 represent distinct prion strains. To answer this question, brain homogenates from sCJDVV cases harboring each of the three resPrP D profiles, were inoculated to transgenic (Tg) mice expressing the human PrP-129M or PrP-129V genotypes. We found that T1 20 and T1 21 were faithfully replicated in Tg129V mice. Electrophoretic profile and incubation period of mice challenged with T1 21−20 resembled those of mice inoculated with T1 21 and T1 20 , respectively. As in sCJDVV1, Tg129V mice challenged with T1 21 and T1 20 generated virtually undistinguishable histotypes. In Tg129M mice, T1 21 was not replicated while T1 20 and T1 21−20 generated a ~ 21–20 kDa doublet after lengthier incubation periods. On second passage, Tg129M mice incubation periods and regional PrP accumulation significantly differed in T1 20 and T1 21−20 challenged mice. Combined, these data indicate that T1 21 and T1 20 resPrP D represent distinct human prion strains associated with partially overlapping histotypes.

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 distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.624
Threshold uncertainty score0.519

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0010.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

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 head0.030
GPT teacher head0.289
Teacher spread0.259 · 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 teacher head, 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

Citations28
Published2021
Admission routes1
Has abstractyes

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