Evidence map›Paper›PMID 35294901›Full record

ArticleNeuron2022

Loss of mouse Stmn2 function causes motor neuropathy.

Irune Guerra San Juan, Leslie A Nash, Kevin S Smith, Marcel F Leyton-Jaimes, Menglu Qian, Joseph R Klim, Francesco Limone, Alexander B Dorr, Alexander Couto, Greta Pintacuda and 9 more

Erratum issuedOpen access · bronzeAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Neuron, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 55 papers.

0numbers the graph read from it
0cells of the map it votes in
55citing papers in PubMed
9.2field-weighted citation impact, top 1% 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

55 citing papers in PubMed, 81 citations in OpenAlex.

  1. Review
  2. Article
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  10. bioRxiv : the preprint server for biology · 2025
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  15. Article
  16. Review
  17. Stathmin-2 enhances motor axon regeneration after injury independent of its binding to tubulin.Proceedings of the National Academy of Sciences of the United States of America · 2025
    Article
  18. Targets and Gene Therapy of ALS (Part 1).International journal of molecular sciences · 2025
    Review
  19. Context-dependent Interactors Regulate TDP-43 Dysfunction in ALS/FTLD.bioRxiv : the preprint server for biology · 2025
    Article
  20. TDP-43 Cryptic RNAs in Perry Syndrome: Differences across Brain Regions and TDP-43 Proteinopathies.Movement disorders : official journal of the Movement Disorder Society · 2025
    Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

19 authors at 3 institutions in 2 countries.

Irune Guerra San JuanDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA; Stanley Center for Psychiatric Research, Broad Institute of MIT and Harvard, Cambridge, MA 02142, USA; Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA; Department of Functional Genomics, Center for Neurogenomics and Cognitive Research, Vrije Universiteit Amsterdam, 1081 HV Amsterdam, the Netherlands; Human Genetics, Amsterdam University Medical Center, 1081 HV Amsterdam, the Netherlands.
Leslie A NashDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA; Stanley Center for Psychiatric Research, Broad Institute of MIT and Harvard, Cambridge, MA 02142, USA; Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA.
Kevin S SmithDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA; Stanley Center for Psychiatric Research, Broad Institute of MIT and Harvard, Cambridge, MA 02142, USA; Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA.
Marcel F Leyton-JaimesDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA; Stanley Center for Psychiatric Research, Broad Institute of MIT and Harvard, Cambridge, MA 02142, USA; Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA.
Menglu QianDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA; Stanley Center for Psychiatric Research, Broad Institute of MIT and Harvard, Cambridge, MA 02142, USA; Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA.
Joseph R KlimDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA; Stanley Center for Psychiatric Research, Broad Institute of MIT and Harvard, Cambridge, MA 02142, USA; Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA.
Francesco LimoneDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA; Stanley Center for Psychiatric Research, Broad Institute of MIT and Harvard, Cambridge, MA 02142, USA; Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA; Leiden University Medical Center, 2333 ZA Leiden, the Netherlands.
Alexander B DorrDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA; Stanley Center for Psychiatric Research, Broad Institute of MIT and Harvard, Cambridge, MA 02142, USA; Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA.
Alexander CoutoDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA; Stanley Center for Psychiatric Research, Broad Institute of MIT and Harvard, Cambridge, MA 02142, USA; Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA.
Greta PintacudaStanley Center for Psychiatric Research, Broad Institute of MIT and Harvard, Cambridge, MA 02142, USA.
Brian J JosephDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA; Stanley Center for Psychiatric Research, Broad Institute of MIT and Harvard, Cambridge, MA 02142, USA; Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA.
D Eric WhisenantDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA.
Caroline NobleDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA.
Veronika MelnikDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA.
Deirdre PotterDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA.
Amie HolmesDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA.
Aaron BurberryDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA; Stanley Center for Psychiatric Research, Broad Institute of MIT and Harvard, Cambridge, MA 02142, USA; Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA; Department of Pathology, Case Western Reserve University, Cleveland, OH 44106, USA.
Matthijs VerhageDepartment of Functional Genomics, Center for Neurogenomics and Cognitive Research, Vrije Universiteit Amsterdam, 1081 HV Amsterdam, the Netherlands; Human Genetics, Amsterdam University Medical Center, 1081 HV Amsterdam, the Netherlands.
Kevin EgganDepartment of Stem Cell and Regenerative Biology, Harvard University, Cambridge, MA 02138, USA; Stanley Center for Psychiatric Research, Broad Institute of MIT and Harvard, Cambridge, MA 02142, USA; Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA. Electronic address: kevin.eggan@bmrn.com.
Broad Institute · USHarvard University · USAmsterdam University Medical Centers · NL

Funding

C9ORF72 in Motor System Biology and ALSR01NS089742 · NINDS · HARVARD UNIVERSITY · PI EGGAN, KEVIN C · 2014 to 2018
$2.3M
Exploring Mechanisms of Pathogenicity in C9ORF72 Frontotemporal Dementia and Amyotrophic Lateral SclerosisK99AG057808 · NIA · HARVARD UNIVERSITY · PI BURBERRY, AARON · 2018 to 2019
$217k
NIA NIH HHS K99 AG057808NINDS NIH HHS R01 NS089742
6 · The paper itself

Abstract

Amyotrophic lateral sclerosis (ALS) is characterized by motor neuron degeneration accompanied by aberrant accumulation and loss of function of the RNA-binding protein TDP43. Thus far, it remains unresolved to what extent TDP43 loss of function directly contributes to motor system dysfunction. Here, we employed gene editing to find whether the mouse ortholog of the TDP43-regulated gene STMN2 has an important function in maintaining the motor system. Both mosaic founders and homozygous loss-of-function Stmn2 mice exhibited neuromuscular junction denervation and fragmentation, resulting in muscle atrophy and impaired motor behavior, accompanied by an imbalance in neuronal microtubule dynamics in the spinal cord. The introduction of human STMN2 through BAC transgenesis was sufficient to rescue the motor phenotypes observed in Stmn2 mutant mice. Collectively, our results demonstrate that disrupting the ortholog of a single TDP43-regulated RNA is sufficient to cause substantial motor dysfunction, indicating that disruption of TDP43 function is likely a contributor to ALS.

Indexed as

Amyotrophic Lateral SclerosisStathminAnimalsDisease Models, AnimalHomozygoteMiceMice, TransgenicMotor NeuronsNeuromuscular JunctionStathminStmn2 protein, mouseALSCRISPRmicrotubulesmotor neuronmotor neuropathyneuromuscular junctionSCG10stathmin 2TARDBPTDP43

Identifiers

PMID35294901
PMCPMC9119928
OpenAlexW4220953393

What OpenQuestion holds

Textmetadata
LicenceTDM
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.