ArticleDisease models & mechanisms2014
Age-dependent pattern of cerebellar susceptibility to bilirubin neurotoxicity in vivo in mice.
Article in Disease models & mechanisms, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 29 papers.
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Who cites it
29 citing papers in PubMed, 41 citations in OpenAlex.
- Transcriptomic and Proteomic Changes in the Brain Along with Increasing Phenotypic Severity in a Rat Model of Neonatal Hyperbilirubinemia.International journal of molecular sciences · 2025Article
- A myotropic AAV vector combined with skeletal muscleMolecular therapy. Methods & clinical development · 2025Article
- Maternal linoleic acid-rich diet ameliorates bilirubin neurotoxicity in offspring mice.Cell death discovery · 2024Article
- Cytomegalovirus infection lengthens the cell cycle of granule cell precursors during postnatal cerebellar development.JCI insight · 2024Article
- Developing and evaluating a predictive model for neonatal hyperbilirubinemia based onFrontiers in pediatrics · 2024Article
- CRISPR-Cas9-mediated somatic correction of a one-base deletion in the Ugt1a gene ameliorates hyperbilirubinemia in Crigler-Najjar syndrome mice.Molecular therapy. Methods & clinical development · 2023Article
- Lipid nanoparticle-encapsulated mRNA therapy corrects serum total bilirubin level in Crigler-Najjar syndrome mouse model.Molecular therapy. Methods & clinical development · 2023Article
- Models of bilirubin neurological damage: lessons learned and new challenges.Pediatric research · 2023Review
- Effects of bilirubin on the development and electrical activity of neural circuits.Frontiers in cellular neuroscience · 2023Review
- Activation of Alternative Bilirubin Clearance Pathways Partially Reduces Hyperbilirubinemia in a Mouse Model Lacking Functional Ugt1a1 Activity.International journal of molecular sciences · 2022Article
- Bilirubin Encephalopathy.Current neurology and neuroscience reports · 2022Review
- Long-Term Effects of Biliverdin Reductase a Deficiency inAntioxidants (Basel, Switzerland) · 2021Article
- Low efficacy of recombinant SV40 in Ugt1a1-/- mice with severe inherited hyperbilirubinemia.PloS one · 2021Article
- Pathological Significance and Prognostic Roles of Indirect Bilirubin/Albumin Ratio in Hepatic Encephalopathy.Frontiers in medicine · 2021Article
- Microglia Susceptibility to Free Bilirubin Is Age-Dependent.Frontiers in pharmacology · 2020Article
- Experimental models assessing bilirubin neurotoxicity.Pediatric research · 2020Review
- Review of bilirubin neurotoxicity II: preventing and treating acute bilirubin encephalopathy and kernicterus spectrum disorders.Pediatric research · 2020Review
- Coupling AAV-mediated promoterless gene targeting to SaCas9 nuclease to efficiently correct liver metabolic diseases.JCI insight · 2019Article
- cVEMP correlated with imbalance in a mouse model of vestibular disorder.Environmental health and preventive medicine · 2019Article
- LED-phototherapy does not induce oxidative DNA damage in hyperbilirubinemic Gunn rats.Pediatric research · 2019Article
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Authors and funding
6 authors at 2 institutions in 1 country.
Funding
Abstract
Neonatal jaundice is caused by high levels of unconjugated bilirubin. It is usually a temporary condition caused by delayed induction of UGT1A1, which conjugates bilirubin in the liver. To reduce bilirubin levels, affected babies are exposed to phototherapy (PT), which converts toxic bilirubin into water-soluble photoisomers that are readily excreted out. However, in some cases uncontrolled hyperbilirubinemia leads to neurotoxicity. To study the mechanisms of bilirubin-induced neurological damage (BIND) in vivo, we generated a mouse model lacking the Ugt1a1 protein and, consequently, mutant mice developed jaundice as early as 36 hours after birth. The mutation was transferred into two genetic backgrounds (C57BL/6 and FVB/NJ). We exposed mutant mice to PT for different periods and analyzed the resulting phenotypes from the molecular, histological and behavioral points of view. Severity of BIND was associated with genetic background, with 50% survival of C57BL/6‑Ugt1(-/-) mutant mice at postnatal day 5 (P5), and of FVB/NJ-Ugt1(-/-) mice at P11. Life-long exposure to PT prevented cerebellar architecture alterations and rescued neuronal damage in FVB/NJ-Ugt1(-/-) but not in C57BL/6-Ugt1(-/-) mice. Survival of FVB/NJ-Ugt1(-/-) mice was directly related to the extent of PT treatment. PT treatment of FVB/NJ-Ugt1(-/-) mice from P0 to P8 did not prevent bilirubin-induced reduction in dendritic arborization and spine density of Purkinje cells. Moreover, PT treatment from P8 to P20 did not rescue BIND accumulated up to P8. However, PT treatment administered in the time-window P0-P15 was sufficient to obtain full rescue of cerebellar damage and motor impairment in FVB/NJ-Ugt1(-/-) mice. The possibility to modulate the severity of the phenotype by PT makes FVB/NJ-Ugt1(-/-) mice an excellent and versatile model to study bilirubin neurotoxicity, the role of modifier genes, alternative therapies and cerebellar development during high bilirubin conditions.
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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.