Reference - Detail
| RRC ID | 87472 |
|---|---|
| Author | Ujibe K, Kashima M, Kataoka M, Shimada R, Okamoto M, Kobayashi I, Wada S, Matsuda H, Sakamoto A, Hirata H. |
| Title | Deficiency of Werner RecQ-type DNA helicase causes premature malnutrition in zebrafish. |
| Journal | iScience |
| Abstract |
Werner syndrome is a genetic progeria characterized by premature aging symptoms, but its early-onset pathology remains unclear. We generated wrn truncation mutant (wrn-/-) zebrafish using CRISPR/Cas9 and identified two premature mortality phases: 7-21 and 60-90 days post-fertilization (dpf). Time-course transcriptomics revealed two wrn-/- subgroups. One showed the reduced expression of intestinal and pancreatic exocrine genes at 7-9 dpf, while the other maintained normal expression initially but eventually showed reduced pancreatic exocrine genes by 21-35 dpf. The prematurely dying wrn-/- larvae exhibited intestinal villi and pancreatic defects, along with DNA damage, cell-cycle arrest, and apoptosis. They also had lower glycogen, glucose, and fat levels compared to wild-type and late-dying wrn-/- larvae, suggesting malnutrition. Notably, excess feeding partially improved their survival. These findings reveal early pathological features in the zebrafish model of Werner syndrome. |
| Volume | 29(3) |
| Pages | 114760 |
| Published | 2026-3-20 |
| DOI | 10.1016/j.isci.2026.114760 |
| PII | S2589-0042(26)00135-5 |
| PMID | 41732282 |
| PMC | PMC12924724 |
| IF | 4.447 |
| Altmetric score |
オルトメトリクス指標項目
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| The most frequently cited source | X(Twitter) |
| Total number of mentions | 2 |
| Altmetric score changes over past 6months | 0.7 |
| Resource | |
| Zebrafish | wrn agu26 |