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In Vivo Modeling of the Morbid Human Genome using Danio rerio
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Zebra-Sphinx: Modeling Sphingolipidoses in Zebrafish.

Luca Mignani1, Jessica Guerra1, Marzia Corli1

  • 1Unit of Experimental Oncology and Immunology and Zebrafish Facility, Department of Molecular and Translational Medicine, University of Brescia, 25123 Brescia, Italy.

International Journal of Molecular Sciences
|March 11, 2023
PubMed
Summary
This summary is machine-generated.

Sphingolipidoses are genetic metabolic disorders causing substrate buildup in lysosomes. Zebrafish (Danio rerio) offer a valuable in vivo model for studying these diseases and developing new therapies.

Keywords:
gene knockouthereditary diseaselysosomemorpholinosphingolipidzebrafish

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Area of Science:

  • Biochemistry
  • Genetics
  • Developmental Biology

Background:

  • Sphingolipidoses are lysosomal storage diseases caused by genetic defects in sphingolipid metabolism.
  • These disorders lead to substrate accumulation within lysosomes, resulting in a spectrum of clinical presentations from mild to severe.
  • Current therapeutic strategies require enhancement, necessitating the development of improved in vivo models for research.

Purpose of the Study:

  • To review the utility of zebrafish (Danio rerio) as a model organism for studying sphingolipidoses.
  • To explore how zebrafish models can advance understanding of sphingolipidosis pathogenesis.
  • To highlight the potential of zebrafish in identifying novel therapeutic strategies for these metabolic disorders.

Main Methods:

  • Leveraging zebrafish's high genome conservation with humans and advanced genome editing techniques.
  • Utilizing zebrafish's amenability to manipulation and rapid development for disease modeling.
  • Employing lipidomic analyses in zebrafish to confirm the presence of mammalian lipid classes.

Main Results:

  • Zebrafish possess conserved lipid metabolism, enabling the modeling of lipid-related diseases.
  • Genome editing tools facilitate the creation of specific sphingolipidosis models in zebrafish.
  • Lipidomic data processing using mammalian databases is feasible in zebrafish.

Conclusions:

  • Zebrafish represent an innovative and powerful model system for investigating sphingolipidosis pathogenesis.
  • This model holds significant promise for uncovering new therapeutic targets and approaches.
  • Further research using zebrafish can lead to improved patient outcomes for sphingolipid storage diseases.