Understanding transthyretin amyloid fibril formation by in vitro inhibition of amyloid fibril formation, protease sensitivity studies and alanine scanning mutagenesis /

Transthyretin (TTR) amyloid fibril formation can be induced

Bibliographic Details
Main Author: Miroy, Greta Josephine G., 1968-
Format: Thesis Book
Language:English
Published: [Place of publication not identified] : [publisher not identified] ; 1997.
Subjects:
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Description
Summary:Transthyretin (TTR) amyloid fibril formation can be induced
by partial acid denaturation where soluble tetrameric TTR
dissociates into a monomeric intermediate having an altered
tertiary structure. In this dissertation, we demonstrate
that the natural ligand of TTR, thyroxine and a non-natural
ligand, 2,4,6, triiodophenol inhibit TTR fibril formation
efficiently in vitro. Analytical ultracentrifugation and
SDS-PAGE studies demonstrate that suppression of amyloid
fibril formation is due to the stabilization of tetrameric
TTR preventing the formation of the monomeric amyloidogenic
intermediate under conditions where it would normally form.
To further understand the mechanism of amyloid formation the
structure of the monomeric amyloidogenic intermediate was
evaluated by protease sensitivity studies. These studies
demonstrate that there are regions in TTR that are protease
labile under amyloid fibril forming conditions but are stable
to proteolysis under physiological conditions where fibril
formation is not possible. MALDI-TOF mass spectrometry and
N-terminal sequencing of the fragments electroblotted onto a
PVDF membrane have identified the C-strand-loop-D-strand and
the F strand regions to be protease sensitive at acidic pH.
The sensitivity of the C-strand-loop-D-strand region suggests
that the region has become disordered and migrates away from
the P-sheet core of the protein. This conformational change
exposes strand A that can participate in intermolecular P-
sheet interactions facilitating the self-assembly of TTR into
a Phelical amyloid structure. Alanine scanning mutagenesis
within the A strand and Hstrand were carried out to probe the
critical interactions that facilitate the self assembly of
the monomeric amyloidogenic intermediate. Results from this
alanine scanning mutagenesis suggest single amino-acid
replacement within TTR is not sufficient to generate TTR
variants that are capable of forming a monomeric intermediate
but do not self-assemble. In contrast, the stability of
tetrameric TTR is highly sensitive that single amino acid
replacements produced either highly stabilized variants that
do not form amyloid or highly destabilized mutants that have
increased amyloidogenecity. Finally, electrospray mass
spectrometry of recombinant TTR has detected post-
translational modifications in the protein that may mediate
the neurotoxicity of amyloid fibrils.
Item Description:Vita.
"Major Subject: Chemistry".
Physical Description:xvi, 176 leaves : illustrations ; 28 cm.
Issued also on microfiche from University Microfilms Inc.
Bibliography:Includes bibliographical references: pages 164-174.