Evidence map›Paper›PMID 26926995›Full record

ArticleThe Journal of experimental medicine2016

Strictly co-isogenic C57BL/6J-Prnp-/- mice: A rigorous resource for prion science.

Mario Nuvolone, Mario Hermann, Silvia Sorce, Giancarlo Russo, Cinzia Tiberi, Petra Schwarz, Eric Minikel, Despina Sanoudou, Pawel Pelczar, Adriano Aguzzi

Open access · bronzeAbstract read
In one paragraph

Article in The Journal of experimental medicine, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 68 papers.

0numbers the graph read from it
0cells of the map it votes in
68citing papers in PubMed
6.6field-weighted citation impact, top 3% of its field
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

68 citing papers in PubMed, 118 citations in OpenAlex.

  1. Review
  2. PrPStem cell reports · 2026
    Article
  3. Article
  4. Divalent siRNA for prion disease.Nucleic acids research · 2026
    Article
  5. Divalent siRNA for prion disease.bioRxiv : the preprint server for biology · 2026
    Article
  6. Enhanced lysosomal activity prevents infection with PrPThe Journal of biological chemistry · 2026
    Article
  7. Article
  8. Article
  9. Article
  10. Partial Deletion of the Carboxyl-Terminal Signal Sequence of the Cellular Prion Protein Alters Protein Expression via Endoplasmic Reticulum-Associated Degradation.FASEB journal : official publication of the Federation of American Societies for Experimental Biology · 2025
    Article
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  12. Article
  13. Review
  14. Review
  15. Article
  16. Article
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  18. Article
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  20. Article

8 more citing papers are in PubMed but not listed here.

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

10 authors at 4 institutions in 3 countries.

Mario NuvoloneInstitute of Neuropathology, University Hospital of Zurich, 8091 Zurich, Switzerland.
Mario HermannInstitute of Neuropathology, University Hospital of Zurich, 8091 Zurich, Switzerland Institute of Laboratory Animal Science, University of Zurich, 8091 Zurich, Switzerland.
Silvia SorceInstitute of Neuropathology, University Hospital of Zurich, 8091 Zurich, Switzerland.
Giancarlo RussoFunctional Genomics Center Zurich (FGCZ), 8057 Zurich, Switzerland.
Cinzia TiberiInstitute of Neuropathology, University Hospital of Zurich, 8091 Zurich, Switzerland.
Petra SchwarzInstitute of Neuropathology, University Hospital of Zurich, 8091 Zurich, Switzerland.
Eric MinikelPrion Alliance, Cambridge, MA 02139 Broad Institute, Cambridge, MA 02142 Analytical and Translational Genetics Unit, Massachusetts General Hospital, Boston, MA 02114.
Despina SanoudouFourth Department of Internal Medicine, Attikon Hospital, Medical School, University of Athens, 115 27 Athens, Greece.
Pawel PelczarInstitute of Laboratory Animal Science, University of Zurich, 8091 Zurich, Switzerland.
Adriano AguzziInstitute of Neuropathology, University Hospital of Zurich, 8091 Zurich, Switzerland adriano.aguzzi@usz.ch.
University Hospital of Zurich · CHMassachusetts General Hospital · USNational and Kapodistrian University of Athens · GRUniversity of Zurich · CH

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Although its involvement in prion replication and neurotoxicity during transmissible spongiform encephalopathies is undisputed, the physiological role of the cellular prion protein (PrP(C)) remains enigmatic. A plethora of functions have been ascribed to PrP(C) based on phenotypes of Prnp(-/-) mice. However, all currently available Prnp(-/-) lines were generated in embryonic stem cells from the 129 strain of the laboratory mouse and mostly crossed to non-129 strains. Therefore, Prnp-linked loci polymorphic between 129 and the backcrossing strain resulted in systematic genetic confounders and led to erroneous conclusions. We used TALEN-mediated genome editing in fertilized mouse oocytes to create the Zurich-3 (ZH3) Prnp-ablated allele on a pure C57BL/6J genetic background. Genomic, transcriptional, and phenotypic characterization of Prnp(ZH3/ZH3) mice failed to identify phenotypes previously described in non-co-isogenic Prnp(-/-) mice. However, aged Prnp(ZH3/ZH3) mice developed a chronic demyelinating peripheral neuropathy, confirming the crucial involvement of PrP(C) in peripheral myelin maintenance. This new line represents a rigorous genetic resource for studying the role of PrP(C) in physiology and disease.

Indexed as

AnimalsBase SequenceChromosomes, MammalianEndonucleasesFemaleGene DeletionMacrophagesMaleMice, Inbred C57BLMolecular Sequence DataNerve DegenerationOpen Reading FramesPhagocytosisPhenotypePolyradiculoneuropathyPrionsEndonucleasesPrionsRNATrans-Activators

Identifiers

PMID26926995
PMCPMC4813672
OpenAlexW2299638108

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-SA
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.