Evidence map›Paper›PMID 32697780›Full record

ArticlePLoS genetics2020

Developmental loss of neurofibromin across distributed neuronal circuits drives excessive grooming in Drosophila.

Lanikea B King, Tamara Boto, Valentina Botero, Ari M Aviles, Breanna M Jomsky, Chevara Joseph, James A Walker, Seth M Tomchik

Open access · goldAbstract read
In one paragraph

Article in PLoS genetics, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 19 papers.

0numbers the graph read from it
0cells of the map it votes in
19citing papers in PubMed
1.2field-weighted citation impact, top 24% 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

19 citing papers in PubMed, 22 citations in OpenAlex.

  1. Article
  2. Automated identification of ataxia and convulsions in hyperexcitablebioRxiv : the preprint server for biology · 2025
    Article
  3. Article
  4. Neurofibromin Deficiency Alters the Patterning and Prioritization of Motor Behaviors in a State-Dependent Manner.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2025
    Article
  5. Article
  6. Article
  7. Review
  8. Article
  9. Review
  10. Modeling neurodegenerative and neurodevelopmental disorders in theLearning & memory (Cold Spring Harbor, N.Y.) · 2024
    Review
  11. Cells · 2024
    Review
  12. Neurobiology of sleep and circadian rhythms · 2023
    Article
  13. Article
  14. Loss ofThe Journal of neuroscience : the official journal of the Society for Neuroscience · 2022
    Article
  15. Role of nerves in neurofibromatosis type 1-related nervous system tumors.Cellular oncology (Dordrecht, Netherlands) · 2022
    Review
  16. Review
  17. Review
  18. Article
  19. Article
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

8 authors at 2 institutions in 1 country.

Lanikea B KingDepartment of Neuroscience, The Scripps Research Institute, Jupiter, Florida, United States of America.ORCID 0000-0002-2267-997X
Tamara BotoDepartment of Neuroscience, The Scripps Research Institute, Jupiter, Florida, United States of America.ORCID 0000-0002-9974-3714
Valentina BoteroDepartment of Neuroscience, The Scripps Research Institute, Jupiter, Florida, United States of America.ORCID 0000-0002-9744-3929
Ari M AvilesDepartment of Neuroscience, The Scripps Research Institute, Jupiter, Florida, United States of America.
Breanna M JomskyDepartment of Neuroscience, The Scripps Research Institute, Jupiter, Florida, United States of America.ORCID 0000-0002-8505-4002
Chevara JosephDepartment of Neuroscience, The Scripps Research Institute, Jupiter, Florida, United States of America.ORCID 0000-0002-6906-0779
James A WalkerCenter for Genomic Medicine, Massachusetts General Hospital, Harvard Medical School, Cambridge, Massachusetts, United States of America.ORCID 0000-0001-5585-698X
Seth M TomchikDepartment of Neuroscience, The Scripps Research Institute, Jupiter, Florida, United States of America.ORCID 0000-0001-5686-0833
Scripps Research Institute · USHarvard University · US

Funding

Resource Component: Acquisition, maintenance and distribution of Drosophila stocksP40OD018537 · OD · TRUSTEES OF INDIANA UNIVERSITY · PI Annette L. Parks · 2014 to 2026
$13.5M
Mechanisms of Nf1 Pathophysiology Underlying HyperactivityR01NS097237 · NINDS · UNIVERSITY OF IOWA · PI TOMCHIK, SETH M · 2017 to 2021
$2.4M
Functional analysis of pathogenic mutations in Neurofibromatosis Type-1 (NF1)R21NS096402 · NINDS · MASSACHUSETTS GENERAL HOSPITAL · PI WALKER, JAMES ANTHONY · 2016 to 2017
$465k
NIH HHS P40 OD018537NINDS NIH HHS R01 NS097237NINDS NIH HHS R21 NS096402
6 · The paper itself

Abstract

Neurofibromatosis type 1 is a monogenetic disorder that predisposes individuals to tumor formation and cognitive and behavioral symptoms. The neuronal circuitry and developmental events underlying these neurological symptoms are unknown. To better understand how mutations of the underlying gene (NF1) drive behavioral alterations, we have examined grooming in the Drosophila neurofibromatosis 1 model. Mutations of the fly NF1 ortholog drive excessive grooming, and increased grooming was observed in adults when Nf1 was knocked down during development. Furthermore, intact Nf1 Ras GAP-related domain signaling was required to maintain normal grooming. The requirement for Nf1 was distributed across neuronal circuits, which were additive when targeted in parallel, rather than mapping to discrete microcircuits. Overall, these data suggest that broadly-distributed alterations in neuronal function during development, requiring intact Ras signaling, drive key Nf1-mediated behavioral alterations. Thus, global developmental alterations in brain circuits/systems function may contribute to behavioral phenotypes in neurofibromatosis type 1.

Indexed as

AnimalsCognitionDisease Models, AnimalDrosophila melanogasterDrosophila ProteinsEmbryonic DevelopmentEmbryo, NonmammalianGene Knockdown TechniquesGroomingHumansMutationNerve Tissue ProteinsNeurofibromatosis 1Neuronsras GTPase-Activating ProteinsDrosophila ProteinsNerve Tissue ProteinsNF1 protein, Drosophilaras GTPase-Activating Proteins

Identifiers

PMID32697780
PMCPMC7398555
OpenAlexW3044433718

What OpenQuestion holds

Textmetadata
LicenceCC BY
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.