ArticleCell research2015
Mutation of kri1l causes definitive hematopoiesis failure via PERK-dependent excessive autophagy induction.
Article in Cell research, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 23 papers.
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
23 citing papers in PubMed, 38 citations in OpenAlex.
- Auranofin potentiates linezolid activity against MRSA by disrupting redox homeostasis and inhibiting SarA-mediated virulence and biofilm.Virulence · 2026Article
- Succinate supplementation ameliorates musculoskeletal defects caused by PLOD3 mutations in a BCARD syndrome model.Genome medicine · 2026Article
- Knockdown-Induced Fasting Phenotypes in Flatworms: Insights into Underlying Mechanisms of Feeding Behavior.International journal of molecular sciences · 2025Article
- Bi-allelic deleterious variants in SNAPIN, which encodes a retrograde dynein adaptor, cause a prenatal-onset neurodevelopmental disorder.American journal of human genetics · 2025Article
- Autophagy in aquatic animals: mechanisms, implications, and future directions.Frontiers in immunology · 2025Review
- Metabolomic-based analysis reveals bile acid-mediated ovarian failure induced by low temperature in zebrafish.Zoological research · 2024Article
- Mutant kri1l causes abnormal retinal development via cell cycle arrest and apoptosis induction.Cell death discovery · 2024Article
- Distinct roles of core autophagy-related genes in zebrafish definitive hematopoiesis.Autophagy · 2024Article
- Exploring hematopoiesis in zebrafish using forward genetic screening.Experimental & molecular medicine · 2024Review
- The Zinc Transporter SLC39A10 Plays an Essential Role in Embryonic Hematopoiesis.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2023Article
- Quality assurance of hematopoietic stem cells by macrophages determines stem cell clonality.Science (New York, N.Y.) · 2022Article
- Ercc2/Xpd deficiency results in failure of digestive organ growth in zebrafish with elevated nucleolar stress.iScience · 2022Article
- Article
- Selective and competitive functions of the AAR and UPR pathways in stress-induced angiogenesis.Cell discovery · 2021Article
- The Ribosome Biogenesis Factor Ltv1 Is Essential for Digestive Organ Development and Definitive Hematopoiesis in Zebrafish.Frontiers in cell and developmental biology · 2021Article
- Mutation of Gemin5 Causes Defective Hematopoietic Stem/Progenitor Cells Proliferation in Zebrafish Embryonic Hematopoiesis.Frontiers in cell and developmental biology · 2021Article
- NdInternational journal of nanomedicine · 2020Article
- Molecular mechanisms of ferroptosis and its role in cancer therapy.Journal of cellular and molecular medicine · 2019Review
- VCAM-1Nature · 2018Article
- Studying Autophagy in Zebrafish.Cells · 2017Review
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
27 authors at 8 institutions in 2 countries.
Funding
Abstract
Dysregulation of ribosome biogenesis causes human diseases, such as Diamond-Blackfan anemia, del (5q-) syndrome and bone marrow failure. However, the mechanisms of blood disorders in these diseases remain elusive. Through genetic mapping, molecular cloning and mechanism characterization of the zebrafish mutant cas002, we reveal a novel connection between ribosomal dysfunction and excessive autophagy in the regulation of hematopoietic stem/progenitor cells (HSPCs). cas002 carries a recessive lethal mutation in kri1l gene that encodes an essential component of rRNA small subunit processome. We show that Kri1l is required for normal ribosome biogenesis, expansion of definitive HSPCs and subsequent lineage differentiation. Through live imaging and biochemical studies, we find that loss of Kri1l causes the accumulation of misfolded proteins and excessive PERK activation-dependent autophagy in HSPCs. Blocking autophagy but not inhibiting apoptosis by Bcl2 overexpression can fully rescue hematopoietic defects, but not the lethality of kri1l(cas002) embryos. Treatment with autophagy inhibitors (3-MA and Baf A1) or PERK inhibitor (GSK2656157), or knockdown of beclin1 or perk can markedly restore HSPC proliferation and definitive hematopoietic cell differentiation. These results may provide leads for effective therapeutics that benefit patients with anemia or bone marrow failure caused by ribosome disorders.
Indexed as
Identifiers
What OpenQuestion holds
Registered trials
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.