Exploration of the Binding Behavior and Function of Saposin B
Date of Award
August 2018
Degree Type
Dissertation
Degree Name
Doctor of Philosophy (PhD)
Department
Chemistry
Advisor(s)
Robert P. Doyle
Subject Categories
Physical Sciences and Mathematics
Abstract
This thesis seeks to address the binding behavior and functionality of the lysosomal sphingolipid activator protein saposin B (sapB). SapB is critical for the degradation of O-sulfogalactosylceramide by arylsulfatase A and deficiency of sapB leads to a variant form of the fatal lysosomal storage disease Metachromatic leukodystrophy. Discussed in this thesis is the ability of sapB to bind a broader variety of ligands than has been previously shown. SapB is also shown to bind the bisretinoid A2E and prevent its enzymatic and photooxidation, with implications for the pathogenesis of Age-Related Macular Degeneration. Herein, it is also demonstrated that human patients taking the anti-malarial chloroquine have [sapB-chloroquine]complex in their urine, and the implications this has on the continued use of chloroquine treatment are discussed.
Access
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Recommended Citation
Tinklepaugh, Jay, "Exploration of the Binding Behavior and Function of Saposin B" (2018). Dissertations - ALL. 940.
https://surface.syr.edu/etd/940