ArticleDisease models & mechanisms2018
Modeling Niemann-Pick disease type C1 in zebrafish: a robust platform for
Open access · goldAbstract read
In one paragraph
Article in Disease models & mechanisms, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 31 papers, 1 of them a synthesis that pooled it.
0numbers the graph read from it
0cells of the map it votes in
31citing papers in PubMed, 1 pooled it
4.0field-weighted citation impact, top 6% of its field
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.
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.
Who cites it
31 citing papers in PubMed, 1 synthesis or guideline pooled it, 44 citations in OpenAlex.
- Pooled it
- Mito-TEMPO improves survival rates inScience advances · 2026Article
- Investigating Psychopharmaceutical Effects on Early Vertebrate Development Using a Zebrafish Model System.Journal of developmental biology · 2025Article
- CRISPR/Cas System: A Powerful Strategy to Improve Monogenic Human Diseases as Therapeutic Delivery; Current Applications and Challenges.Current gene therapy · 2025Review
- A high-cholesterol zebrafish diet promotes hypercholesterolemia and fasting-associated liver steatosis.Journal of lipid research · 2024Article
- Zebra-Sphinx: Modeling Sphingolipidoses in Zebrafish.International journal of molecular sciences · 2023Review
- Severe neurometabolic phenotype inFrontiers in molecular neuroscience · 2023Article
- Current landscape of gene-editing technology in biomedicine: Applications, advantages, challenges, and perspectives.MedComm · 2022Review
- Article
- Article
- RedEfish: Generation of the Polycistronic mScarlet: GSG-T2A: Ttpa Zebrafish Line.Antioxidants (Basel, Switzerland) · 2021Article
- Proteotyping of knockout mouse strains reveals sex- and strain-specific signatures in blood plasma.NPJ systems biology and applications · 2021Article
- A Niemann-pick C1 disease child with BCG-itis: a case report and analysis.BMC pediatrics · 2021Article
- Zebrafish Models of Autosomal Recessive Ataxias.Cells · 2021Review
- The role of Niemann-Pick type C2 in zebrafish embryonic development.Development (Cambridge, England) · 2021Article
- Article
- Zebrafish as an Emerging Model for Dyslipidemia and Associated Diseases.Journal of lipid and atherosclerosis · 2021Review
- Review
- Understanding and Treating Niemann-Pick Type C Disease: Models Matter.International journal of molecular sciences · 2020Review
- Visualisation of cholesterol and ganglioside GM1 in zebrafish models of Niemann-Pick type C disease and Smith-Lemli-Opitz syndrome using light sheet microscopy.Histochemistry and cell biology · 2020Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
11 authors at 1 institution in 1 country.
Wei-Chia TsengSection on Molecular Dysmorphology, Division of Translational Research, Eunice Kennedy Shriver National Institute of Child Health and Human Development, National Institutes of Health, Department of Health and Human Services, Bethesda, MD 20892, USA.ORCID 0000-0002-7172-2102
Hannah E LoebSection on Molecular Dysmorphology, Division of Translational Research, Eunice Kennedy Shriver National Institute of Child Health and Human Development, National Institutes of Health, Department of Health and Human Services, Bethesda, MD 20892, USA.
Wuhong PeiTranslational and Functional Genomics Branch, National Human Genome Research Institute, National Institutes of Health, Department of Health and Human Services, Bethesda, MD 20892, USA.
Chon-Hwa Tsai-MorrisZebrafish Core, Eunice Kennedy Shriver National Institute of Child Health and Human Development, National Institutes of Health, Department of Health and Human Services, Bethesda, MD 20892, USA.
Lisha XuTranslational and Functional Genomics Branch, National Human Genome Research Institute, National Institutes of Health, Department of Health and Human Services, Bethesda, MD 20892, USA.ORCID 0000-0002-2567-7785
Celine V CluzeauSection on Molecular Dysmorphology, Division of Translational Research, Eunice Kennedy Shriver National Institute of Child Health and Human Development, National Institutes of Health, Department of Health and Human Services, Bethesda, MD 20892, USA.ORCID 0000-0002-7791-6223
Christopher A WassifSection on Molecular Dysmorphology, Division of Translational Research, Eunice Kennedy Shriver National Institute of Child Health and Human Development, National Institutes of Health, Department of Health and Human Services, Bethesda, MD 20892, USA.ORCID 0000-0002-2524-1420
Benjamin FeldmanZebrafish Core, Eunice Kennedy Shriver National Institute of Child Health and Human Development, National Institutes of Health, Department of Health and Human Services, Bethesda, MD 20892, USA.ORCID 0000-0001-8880-3454
Shawn M BurgessTranslational and Functional Genomics Branch, National Human Genome Research Institute, National Institutes of Health, Department of Health and Human Services, Bethesda, MD 20892, USA.ORCID 0000-0003-1147-0596
William J PavanGenetic Disease Research Branch, National Human Genome Research Institute, National Institutes of Health, Department of Health and Human Services, Bethesda, MD 20892, USA.ORCID 0000-0001-8281-5120
Forbes D PorterSection on Molecular Dysmorphology, Division of Translational Research, Eunice Kennedy Shriver National Institute of Child Health and Human Development, National Institutes of Health, Department of Health and Human Services, Bethesda, MD 20892, USA fdporter@mail.nih.gov.ORCID 0000-0001-9397-0046
National Institutes of Health · US
Funding
Zebrafish targeted mutagenesis and functional genomicsZIAHG000183 · NHGRI · NATIONAL HUMAN GENOME RESEARCH INSTITUTE · PI BURGESS, SHAWN M · 2009 to 2025
$16.7MHearing regeneration in zebrafishZIAHG200386 · NHGRI · NATIONAL HUMAN GENOME RESEARCH INSTITUTE · PI BURGESS, SHAWN M · 2012 to 2025
$12.5MInborn Errors of Cholesterol SynthesisZIAHD000139 · NICHD · EUNICE KENNEDY SHRIVER NATIONAL INSTITUTE OF CHILD HEALTH & HUMAN DEVELOPMENT · PI PORTER, FORBES · 2009 to 2019
$11.7MGene Function, Expression and Regulation in ZebrafishZICHD008921 · NICHD · EUNICE KENNEDY SHRIVER NATIONAL INSTITUTE OF CHILD HEALTH & HUMAN DEVELOPMENT · PI FELDMAN, BENJAMIN · 2012 to 2023
$6.1MTHE USE OF THE MOUSE NPC MODEL TO CLONE THE HUMAN DISEASE GENEZIAHG000068 · NHGRI · NATIONAL HUMAN GENOME RESEARCH INSTITUTE · PI PAVAN, WILLIAM J · 2009 to 2023
$6.0MClinical Investigations of Niemann-Pick Disease, type CZIAHD008824 · NICHD · EUNICE KENNEDY SHRIVER NATIONAL INSTITUTE OF CHILD HEALTH & HUMAN DEVELOPMENT · PI PORTER, FORBES · 2009 to 2019
$5.2MIntramural NIH HHS ZIA HD000139
Abstract
Niemann-Pick disease type C1 (NPC1) is a rare autosomal recessive lysosomal storage disease primarily caused by mutations in
Indexed as
Drug Evaluation, Preclinical2-Hydroxypropyl-beta-cyclodextrinAllelesAnimalsBase SequenceBrainCholesterolDisease Models, AnimalLarvaLiverLiver DiseasesMembrane ProteinsMutationNeuronsNiemann-Pick C1 ProteinNiemann-Pick Disease, Type C2-Hydroxypropyl-beta-cyclodextrinCholesterolMembrane ProteinsNiemann-Pick C1 ProteinNPC1 protein, zebrafishZebrafish ProteinsDrug screeningLiver diseaseLysosomal storage disorderNeurodegenerationNiemann-Pick disease type C1Zebrafish
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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.