A Primer Genetic Toolkit for Exploring Mitochondrial Biology and Disease Using Zebrafish Article Swipe
Ankit Sabharwal
,
Jarryd M. Campbell
,
Tanya L. Schwab
,
Zachary WareJoncas
,
Mark Wishman
,
Hirotaka Ata
,
Wiebin Liu
,
Noriko Ichino
,
Danielle E. Hunter
,
Jake D. Bergren
,
Mark D. Urban
,
Rhianna Urban
,
Shannon R. Holmberg
,
Bibekananda Kar
,
Alex R. Cook
,
Yonghe Ding
,
Xiaolei Xu
,
Karl J. Clark
,
Stephen C. Ekker
·
YOU?
·
· 2022
· Open Access
·
· DOI: https://doi.org/10.3390/genes13081317
YOU?
·
· 2022
· Open Access
·
· DOI: https://doi.org/10.3390/genes13081317
Mitochondria are a dynamic eukaryotic innovation that play diverse roles in biology and disease. The mitochondrial genome is remarkably conserved in all vertebrates, encoding the same 37-gene set and overall genomic structure, ranging from 16,596 base pairs (bp) in the teleost zebrafish (Danio rerio) to 16,569 bp in humans. Mitochondrial disorders are amongst the most prevalent inherited diseases, affecting roughly 1 in every 5000 individuals. Currently, few effective treatments exist for those with mitochondrial ailments, representing a major unmet patient need. Mitochondrial dysfunction is also a common component of a wide variety of other human illnesses, ranging from neurodegenerative disorders such as Huntington’s disease and Parkinson’s disease to autoimmune illnesses such as multiple sclerosis and rheumatoid arthritis. The electron transport chain (ETC) component of mitochondria is critical for mitochondrial biology and defects can lead to many mitochondrial disease symptoms. Here, we present a publicly available collection of genetic mutants created in highly conserved, nuclear-encoded mitochondrial genes in Danio rerio. The zebrafish system represents a potentially powerful new opportunity for the study of mitochondrial biology and disease due to the large number of orthologous genes shared with humans and the many advanced features of this model system, from genetics to imaging. This collection includes 15 mutant lines in 13 different genes created through locus-specific gene editing to induce frameshift or splice acceptor mutations, leading to predicted protein truncation during translation. Additionally, included are 11 lines created by the random insertion of the gene-breaking transposon (GBT) protein trap cassette. All these targeted mutant alleles truncate conserved domains of genes critical to the proper function of the ETC or genes that have been implicated in human mitochondrial disease. This collection is designed to accelerate the use of zebrafish to study many different aspects of mitochondrial function to widen our understanding of their role in biology and human disease.
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Metadata
- Type
- article
- Language
- en
- Landing Page
- https://doi.org/10.3390/genes13081317
- https://www.mdpi.com/2073-4425/13/8/1317/pdf?version=1661399700
- OA Status
- gold
- Cited By
- 12
- References
- 98
- Related Works
- 10
- OpenAlex ID
- https://openalex.org/W4287009935
All OpenAlex metadata
Raw OpenAlex JSON
- OpenAlex ID
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https://openalex.org/W4287009935Canonical identifier for this work in OpenAlex
- DOI
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https://doi.org/10.3390/genes13081317Digital Object Identifier
- Title
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A Primer Genetic Toolkit for Exploring Mitochondrial Biology and Disease Using ZebrafishWork title
- Type
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articleOpenAlex work type
- Language
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enPrimary language
- Publication year
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2022Year of publication
- Publication date
-
2022-07-23Full publication date if available
- Authors
-
Ankit Sabharwal, Jarryd M. Campbell, Tanya L. Schwab, Zachary WareJoncas, Mark Wishman, Hirotaka Ata, Wiebin Liu, Noriko Ichino, Danielle E. Hunter, Jake D. Bergren, Mark D. Urban, Rhianna Urban, Shannon R. Holmberg, Bibekananda Kar, Alex R. Cook, Yonghe Ding, Xiaolei Xu, Karl J. Clark, Stephen C. EkkerList of authors in order
- Landing page
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https://doi.org/10.3390/genes13081317Publisher landing page
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https://www.mdpi.com/2073-4425/13/8/1317/pdf?version=1661399700Direct link to full text PDF
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-
YesWhether a free full text is available
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goldOpen access status per OpenAlex
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https://www.mdpi.com/2073-4425/13/8/1317/pdf?version=1661399700Direct OA link when available
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Zebrafish, Primer (cosmetics), Biology, Mitochondrial DNA, Computational biology, Genetics, Evolutionary biology, Gene, Chemistry, Organic chemistryTop concepts (fields/topics) attached by OpenAlex
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12Total citation count in OpenAlex
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2025: 6, 2024: 2, 2023: 2, 2022: 1, 2021: 1Per-year citation counts (last 5 years)
- References (count)
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98Number of works referenced by this work
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10Other works algorithmically related by OpenAlex
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| abstract_inverted_index.cassette. | 246 |
| abstract_inverted_index.component | 87, 122 |
| abstract_inverted_index.conserved | 19, 253 |
| abstract_inverted_index.different | 208, 288 |
| abstract_inverted_index.diseases, | 57 |
| abstract_inverted_index.disorders | 50, 99 |
| abstract_inverted_index.effective | 67 |
| abstract_inverted_index.illnesses | 109 |
| abstract_inverted_index.inherited | 56 |
| abstract_inverted_index.insertion | 238 |
| abstract_inverted_index.predicted | 224 |
| abstract_inverted_index.prevalent | 55 |
| abstract_inverted_index.sclerosis | 113 |
| abstract_inverted_index.symptoms. | 138 |
| abstract_inverted_index.transport | 119 |
| abstract_inverted_index.zebrafish | 41, 160, 284 |
| abstract_inverted_index.Currently, | 65 |
| abstract_inverted_index.accelerate | 280 |
| abstract_inverted_index.arthritis. | 116 |
| abstract_inverted_index.autoimmune | 108 |
| abstract_inverted_index.collection | 145, 201, 276 |
| abstract_inverted_index.conserved, | 152 |
| abstract_inverted_index.eukaryotic | 4 |
| abstract_inverted_index.frameshift | 217 |
| abstract_inverted_index.illnesses, | 95 |
| abstract_inverted_index.implicated | 270 |
| abstract_inverted_index.innovation | 5 |
| abstract_inverted_index.mutations, | 221 |
| abstract_inverted_index.remarkably | 18 |
| abstract_inverted_index.represents | 162 |
| abstract_inverted_index.rheumatoid | 115 |
| abstract_inverted_index.structure, | 31 |
| abstract_inverted_index.transposon | 242 |
| abstract_inverted_index.treatments | 68 |
| abstract_inverted_index.truncation | 226 |
| abstract_inverted_index.dysfunction | 82 |
| abstract_inverted_index.opportunity | 167 |
| abstract_inverted_index.orthologous | 182 |
| abstract_inverted_index.potentially | 164 |
| abstract_inverted_index.Mitochondria | 0 |
| abstract_inverted_index.individuals. | 64 |
| abstract_inverted_index.mitochondria | 124 |
| abstract_inverted_index.representing | 75 |
| abstract_inverted_index.translation. | 228 |
| abstract_inverted_index.vertebrates, | 22 |
| abstract_inverted_index.Additionally, | 229 |
| abstract_inverted_index.Mitochondrial | 49, 81 |
| abstract_inverted_index.Parkinson’s | 105 |
| abstract_inverted_index.gene-breaking | 241 |
| abstract_inverted_index.mitochondrial | 15, 73, 128, 136, 154, 172, 273, 291 |
| abstract_inverted_index.understanding | 296 |
| abstract_inverted_index.Huntington’s | 102 |
| abstract_inverted_index.locus-specific | 212 |
| abstract_inverted_index.nuclear-encoded | 153 |
| abstract_inverted_index.neurodegenerative | 98 |
| cited_by_percentile_year.max | 99 |
| cited_by_percentile_year.min | 89 |
| corresponding_author_ids | https://openalex.org/A5044571120 |
| countries_distinct_count | 1 |
| institutions_distinct_count | 19 |
| corresponding_institution_ids | https://openalex.org/I1330342723, https://openalex.org/I2802423016 |
| citation_normalized_percentile.value | 0.75334061 |
| citation_normalized_percentile.is_in_top_1_percent | False |
| citation_normalized_percentile.is_in_top_10_percent | False |