Evidence map›Paper›PMID 31669185›Full record

ArticleProgress in neurobiology2020

Neurofilament-lysosomal genetic intersections in the cortical network of stuttering.

Claudia Benito-Aragón, Ricardo Gonzalez-Sarmiento, Thomas Liddell, Ibai Diez, Federico d'Oleire Uquillas, Laura Ortiz-Terán, Elisenda Bueichekú, Ho Ming Chow, Soo-Eun Chang, Jorge Sepulcre

Abstract read
In one paragraph

Article in Progress in neurobiology, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 23 papers.

0numbers the graph read from it
0cells of the map it votes in
23citing papers in PubMed
–field-weighted citation impact
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

23 citing papers in PubMed.

  1. Article
  2. Review
  3. Unraveling Stuttering Through a Multi-Omics Lens.Life (Basel, Switzerland) · 2025
    Article
  4. Review
  5. Article
  6. Article
  7. Article
  8. Article
  9. Stuttering as a spectrum disorder: A hypothesis.Current research in neurobiology · 2023
    Review
  10. Article
  11. Article
  12. Article
  13. Article
  14. Article
  15. Article
  16. Article
  17. Article
  18. Evaluation of recurrentAdvanced genetics (Hoboken, N.J.) · 2021
    Article
  19. Article
  20. Review
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.

Claudia Benito-AragónGordon Center for Medical Imaging, Department of Radiology, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA; University of Navarra School of Medicine, University of Navarra, Pamplona, Navarra, Spain.
Ricardo Gonzalez-SarmientoGordon Center for Medical Imaging, Department of Radiology, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA; University of Navarra School of Medicine, University of Navarra, Pamplona, Navarra, Spain.
Thomas LiddellGordon Center for Medical Imaging, Department of Radiology, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA; University of Exeter, Exeter, England, UK.
Ibai DiezGordon Center for Medical Imaging, Department of Radiology, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA; Neurotechnology Laboratory, Tecnalia Health Department, Tecnalia, Derio, Basque Country, Spain.
Federico d'Oleire UquillasDepartment of Neurology, Massachusetts General Hospital, Harvard Medical School, Boston, Massachusetts, USA.
Laura Ortiz-TeránGordon Center for Medical Imaging, Department of Radiology, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA; Department of Radiology, Brigham and Women's Hospital, Harvard Medical School, Boston, MA, USA.
Elisenda BueichekúGordon Center for Medical Imaging, Department of Radiology, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA; Neuropsychology and Functional Neuroimaging Group, Department of Basic Psychology, Universitat Jaume I, Castellón, Spain.
Ho Ming ChowDepartment of Psychiatry, University of Michigan, Michigan, USA; Katzin Diagnostic and Research PET/MRI Center, Nemours/Alfred I. duPont Hospital for Children, Wilmington, DE, USA.
Soo-Eun ChangDepartment of Psychiatry, University of Michigan, Michigan, USA; Cognitive Imaging Research Center, Department of Radiology, Michigan State University, East Lansing, MI, USA.
Jorge SepulcreGordon Center for Medical Imaging, Department of Radiology, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA; Athinoula A. Martinos Center for Biomedical Imaging, Department of Radiology, Massachusetts General Hospital and Harvard Medical School, Charlestown, MA, USA. Electronic address: sepulcre@nmr.mgh.harvard.edu.

Funding

Sexual dimorphism of neural development underlying childhood stutteringR01DC011277 · NIDCD · UNIVERSITY OF MICHIGAN AT ANN ARBOR · PI Soo-Eun Chang · 2010 to 2026
$8.2M
Postgraduate Training Program in Medical Imaging (PTPMI)T32EB013180 · NIBIB · MASSACHUSETTS GENERAL HOSPITAL · PI Ciprian Catana, Pedram Heidari · 2011 to 2026
$3.7M
In Vivo Visualization of TAU and Amyloid-Beta Networks for Early AD DetectionK23EB019023 · NIBIB · MASSACHUSETTS GENERAL HOSPITAL · PI SEPULCRE, JORGE · 2014 to 2018
$810k
Imaging genetics study of twins who stutterR21DC015312 · NIDCD · UNIVERSITY OF MICHIGAN AT ANN ARBOR · PI CHANG, SOO-EUN · 2017 to 2018
$440k
NIBIB NIH HHS K23 EB019023NIBIB NIH HHS T32 EB013180NIDCD NIH HHS R01 DC011277NIDCD NIH HHS R21 DC015312
6 · The paper itself

Abstract

The neurobiological underpinnings of stuttering, a speech disorder characterized by disrupted speech fluency, remain unclear. While recent developments in the field have afforded researchers the ability to pinpoint several genetic profiles associated with stuttering, how these specific genetic backgrounds impact neuronal circuits and how they generate or facilitate the emergence of stuttered speech remains unknown. In this study, we identified the large-scale cortical network that characterizes stuttering using functional connectivity MRI and graph theory. We performed a spatial similarity analysis that examines whether the topology of the stuttering cortical network intersects with genetic expression levels of previously reported genes for stuttering from the protein-coding transcriptome data of the Allen Human Brain Atlas. We found that GNPTG - a gene involved in the mannose-6-phosphate lysosomal targeting pathways - was significantly co-localized with the stuttering cortical network. An enrichment analysis demonstrated that the genes identified with the stuttering cortical network shared a significantly overrepresented biological functionality of Neurofilament Cytoskeleton Organization (NEFH, NEFL and INA). The relationship between lysosomal pathways, cytoskeleton organization, and stuttering, was investigated by comparing the genetic interactome between GNPTG and the neurofilament genes implicated in the current study. We found that genes of the interactome network, including CDK5, SNCA, and ACTB, act as functional links between lysosomal and neurofilament genes. These findings support the notion that stuttering is due to a lysosomal dysfunction, which has deleterious effects on the neurofilament organization of the speech neuronal circuits. They help to elucidate the intriguing, unsolved link between lysosomal mutations and the presence of stuttering.

Indexed as

Cerebral CortexConnectomeNerve NetStutteringTranscriptomeAtlases as TopicHumansLysosomesMagnetic Resonance ImagingMeta-Analysis as TopicNeurofilament ProteinsTransferases (Other Substituted Phosphate Groups)GNPTG protein, humanNeurofilament ProteinsTransferases (Other Substituted Phosphate Groups)Cortical networkGeneticsLysosomalNeurofilamentStuttering

Identifiers

PMID31669185
PMCPMC6938554

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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.