Evidence map›Paper›PMID 26711119›Full record

ArticleNeuron2016

Congenital Nystagmus Gene FRMD7 Is Necessary for Establishing a Neuronal Circuit Asymmetry for Direction Selectivity.

Keisuke Yonehara, Michele Fiscella, Antonia Drinnenberg, Federico Esposti, Stuart Trenholm, Jacek Krol, Felix Franke, Brigitte Gross Scherf, Akos Kusnyerik, Jan Müller and 9 more

Open access · bronzeAbstract read
In one paragraph

Article in Neuron, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 96 papers.

0numbers the graph read from it
0cells of the map it votes in
96citing papers in PubMed
6.5field-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

96 citing papers in PubMed, 165 citations in OpenAlex.

  1. Dopamine suppresses pathological retinal oscillations and enhances the signal-to-noise ratio.Proceedings of the National Academy of Sciences of the United States of America · 2026
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  8. Brain communications · 2026
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  14. Diverse and dynamic influences of saccades on visual representations in the mouse superior colliculus.Proceedings of the National Academy of Sciences of the United States of America · 2025
    Article
  15. Article
  16. Article
  17. Article
  18. Article
  19. Article
  20. Article

36 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

19 authors at 6 institutions in 2 countries.

Keisuke YoneharaNeural Circuit Laboratories, Friedrich Miescher Institute for Biomedical Research, Maulbeerstrasse 66, 4058 Basel, Switzerland.
Michele FiscellaBio Engineering Laboratory, Department of Biosystems Science and Engineering of ETH Zurich, Mattenstrasse 26, 4058 Basel, Switzerland.
Antonia DrinnenbergNeural Circuit Laboratories, Friedrich Miescher Institute for Biomedical Research, Maulbeerstrasse 66, 4058 Basel, Switzerland; University of Basel, Petersplatz 1, 4003 Basel, Switzerland.
Federico EspostiNeural Circuit Laboratories, Friedrich Miescher Institute for Biomedical Research, Maulbeerstrasse 66, 4058 Basel, Switzerland.
Stuart TrenholmNeural Circuit Laboratories, Friedrich Miescher Institute for Biomedical Research, Maulbeerstrasse 66, 4058 Basel, Switzerland.
Jacek KrolNeural Circuit Laboratories, Friedrich Miescher Institute for Biomedical Research, Maulbeerstrasse 66, 4058 Basel, Switzerland.
Felix FrankeBio Engineering Laboratory, Department of Biosystems Science and Engineering of ETH Zurich, Mattenstrasse 26, 4058 Basel, Switzerland.
Brigitte Gross ScherfNeural Circuit Laboratories, Friedrich Miescher Institute for Biomedical Research, Maulbeerstrasse 66, 4058 Basel, Switzerland.
Akos KusnyerikDepartment of Ophthalmology, Semmelweis University, Mária u. 39, 1085 Budapest, Hungary.
Jan MüllerBio Engineering Laboratory, Department of Biosystems Science and Engineering of ETH Zurich, Mattenstrasse 26, 4058 Basel, Switzerland.
Arnold SzaboDepartment of Human Morphology and Developmental Biology, Faculty of Medicine, Semmelweis University, Tűzoltó u. 58, 1094 Budapest, Hungary.
Josephine JüttnerNeural Circuit Laboratories, Friedrich Miescher Institute for Biomedical Research, Maulbeerstrasse 66, 4058 Basel, Switzerland.
Francisco CordobaLaboratory and Animal Services, Novartis Institute for Biomedical Research, Fabrikstrasse 28, 4056 Basel, Switzerland.
Ashrithpal Police ReddyNeural Circuit Laboratories, Friedrich Miescher Institute for Biomedical Research, Maulbeerstrasse 66, 4058 Basel, Switzerland.
János NémethDepartment of Ophthalmology, Semmelweis University, Mária u. 39, 1085 Budapest, Hungary.
Zoltán Zsolt NagyDepartment of Ophthalmology, Semmelweis University, Mária u. 39, 1085 Budapest, Hungary.
Francis MunierJules-Gonin Eye Hospital, Avenue de France 15, 1000 Lausanne, Switzerland.
Andreas HierlemannBio Engineering Laboratory, Department of Biosystems Science and Engineering of ETH Zurich, Mattenstrasse 26, 4058 Basel, Switzerland.
Botond RoskaNeural Circuit Laboratories, Friedrich Miescher Institute for Biomedical Research, Maulbeerstrasse 66, 4058 Basel, Switzerland; Department of Ophthalmology, University of Basel, Mittlere Strasse 91, 4031 Basel, Switzerland. Electronic address: botond.roska@fmi.ch.
Friedrich Miescher Institute · CHETH Zurich · CHSemmelweis University · HUHôpital Ophtalmique Jules-Gonin · CHNovartis Institutes for BioMedical ResearchUniversity of Basel · CH

Funding

Transgenic CoreP30DK057521 · NIDDK · MASSACHUSETTS GENERAL HOSPITAL · PI FLOREZ, JOSE CARLOS · 2000 to 2019
$31.4M
European Research Council 267351NIDDK NIH HHS P30 DK057521Swiss National Science Foundation 141801
6 · The paper itself

Abstract

Neuronal circuit asymmetries are important components of brain circuits, but the molecular pathways leading to their establishment remain unknown. Here we found that the mutation of FRMD7, a gene that is defective in human congenital nystagmus, leads to the selective loss of the horizontal optokinetic reflex in mice, as it does in humans. This is accompanied by the selective loss of horizontal direction selectivity in retinal ganglion cells and the transition from asymmetric to symmetric inhibitory input to horizontal direction-selective ganglion cells. In wild-type retinas, we found FRMD7 specifically expressed in starburst amacrine cells, the interneuron type that provides asymmetric inhibition to direction-selective retinal ganglion cells. This work identifies FRMD7 as a key regulator in establishing a neuronal circuit asymmetry, and it suggests the involvement of a specific inhibitory neuron type in the pathophysiology of a neurological disease.

Indexed as

Action PotentialsAmacrine CellsAnimalsCytoskeletal ProteinsMice, TransgenicMotion PerceptionNerve NetNeural InhibitionNystagmus, CongenitalPhotic StimulationRetinaRetinal Ganglion CellsSynapsesVisual PathwaysCytoskeletal ProteinsFRMD7 protein, mouse

Identifiers

PMID26711119
PMCPMC4712192
OpenAlexW2209166030

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
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.