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Iduronate-2-sulfatase interactome: validation by yeast two-hybrid assay
Eliana Benincore-Flórez1, Jorge El-Azaz2, Gabriela Alejandra Solarte1
1Institute for the Study of Inborn Errors of Metabolism, Faculty of Sciences, Pontificia, Universidad Javeriana, Bogotá, Colombia.
Heliyon
|March 14, 2022
Summary
Researchers identified new interactions for the enzyme iduronate-2-sulfatase (IDS), deficient in Hunter syndrome (MPS II). These findings may reveal how IDS functions in the brain and inform new therapies for MPS II.
Area of Science:
- Biochemistry
- Neuroscience
- Genetics
Background:
- Mucopolysaccharidosis type II (MPS II), or Hunter syndrome, is a rare X-linked disorder.
- It stems from a deficiency in the enzyme iduronate-2-sulfatase (IDS).
- IDS deficiency leads to glycosaminoglycan buildup, damaging tissues, especially the central nervous system.
Purpose of the Study:
- To validate previously identified IDS-protein interactions in the mouse brain.
- To investigate potential roles of these interactions in MPS II pathophysiology.
- To explore novel therapeutic strategies for delivering IDS across the blood-brain barrier.
Main Methods:
- Selected and cloned coding regions of 10 candidate genes.
- Performed targeted yeast two-hybrid assays to test protein interactions.
- Analyzed interactions for potential roles in vesicular trafficking and blood-brain barrier transport.
Main Results:
- Identified physical interactions between IDS and LSAMP.
- Identified physical interactions between IDS and SYT1.
- LSAMP and SYT1 interactions suggest roles in IDS vesicular trafficking and blood-brain barrier transcytosis.
Conclusions:
- IDS interacts with LSAMP and SYT1, suggesting roles in cellular homeostasis.
- These interactions may be crucial for transporting IDS across the blood-brain barrier.
- Findings offer insights into MPS II mechanisms and potential therapeutic delivery routes.
