International Journal of
Molecular Sciences
Article
Diphenyl-Methane Based Thyromimetic Inhibitors for
Transthyretin Amyloidosis
Bokyung Kim
1
, Young Ho Ko
2
, Massimiliano Runfola
3
, Simona Rapposelli
3
, Gabriella Ortore
3,
*,
Grazia Chiellini
4,
* and Jin Hae Kim
1,
*
Citation: Kim, B.; Ko, Y.H.; Runfola,
M.; Rapposelli, S.; Ortore, G.;
Chiellini, G.; Kim, J.H.
Diphenyl-Methane Based
Thyromimetic Inhibitors for
Transthyretin Amyloidosis. Int. J. Mol.
Sci. 2021, 22, 3488. https://
doi.org/10.3390/ijms22073488
Academic Editor: Holger Wille
Received: 4 March 2021
Accepted: 25 March 2021
Published: 28 March 2021
Publisher’s Note: MDPI stays neutral
with regard to jurisdictional claims in
published maps and institutional affil-
iations.
Copyright: © 2021 by the authors.
Licensee MDPI, Basel, Switzerland.
This article is an open access article
distributed under the terms and
conditions of the Creative Commons
Attribution (CC BY) license (https://
creativecommons.org/licenses/by/
4.0/).
1
Department of New Biology, Daegu Gyeongbuk Institute of Science & Technology (DGIST),
Daegu 42988, Korea; bkkim@dgist.ac.kr
2
Center for Self-Assembly and Complexity, Institute for Basic Science, Pohang 37673, Korea; yhko@ibs.re.kr
3
Department of Pharmacy, University of Pisa, 56100 Pisa, Italy; massimiliano.runfola@farm.unipi.it (M.R.);
simona.rapposelli@unipi.it (S.R.)
4
Department of Pathology, University of Pisa, 56100 Pisa, Italy
* Correspondence: gabriella.ortore@farm.unipi.it (G.O.); grazia.chiellini@unipi.it (G.C.);
jinhaekim@dgist.ac.kr (J.H.K.)
Abstract: Thyromimetics, whose physicochemical characteristics are analog to thyroid hormones
(THs) and their derivatives, are promising candidates as novel therapeutics for neurodegenerative
and metabolic pathologies. In particular, sobetirome (GC-1), one of the initial halogen-free thy-
romimetics, and newly synthesized IS25 and TG68, with optimized ADME-Tox profile, have recently
attracted attention owing to their superior therapeutic benefits, selectivity, and enhanced permeability.
Here, we further explored the functional capabilities of these thyromimetics to inhibit transthyretin
(TTR) amyloidosis. TTR is a homotetrameric transporter protein for THs, yet it is also responsible
for severe amyloid fibril formation, which is facilitated by tetramer dissociation into non-native
monomers. By combining nuclear magnetic resonance (NMR) spectroscopy, computational simula-
tion, and biochemical assays, we found that GC-1 and newly designed diphenyl-methane-based thy-
romimetics, namely IS25 and TG68, are TTR stabilizers and efficient suppressors of TTR aggregation.
Based on these observations, we propose the novel potential of thyromimetics as a multi-functional
therapeutic molecule for TTR-related pathologies, including neurodegenerative diseases.
Keywords: transthyretin; thyromimetics; sobetirome; TTR amyloidosis; TTR stabilizers; protein ag-
gregation
1. Introduction
Transthyretin (TTR) is an essential transporter of the thyroid hormone (TH) and a
holo-retinol-binding protein. Under physiological conditions, TTR forms a ~55 kDa homote-
trameric complex, on which two hydrophobic pockets are constructed as binding sites for
thyroxine (T
4
)[1,2]. Notably, the quaternary stability of this protein is important not only
for its physiological function but also to suppress its amyloidogenic propensity [3]. In its
native tetrameric state, TTR is a non-amyloidogenic protein, whereas its dissociation into
non-native monomers facilitates aggregation and formation of amyloid fibrils [4]. Due to
the relative predominance of TTR in human serum and cerebrospinal fluid, TTR amyloido-
sis results in detrimental amyloidogenic diseases. For example, senile systemic amyloidosis
is caused by the spontaneous aggregation of wild-type (WT) TTR, while familial amyloid
polyneuropathy or familial amyloid cardiomyopathy is attributed to genetic modifications
of TTR [5]. Indeed, more than 100 mutations have been reported to date, and it was shown
that most pathogenic mutations facilitate fibril formation by reducing the stability of the
TTR tetramer [6]. Recent investigations in the USA estimated that over three percent of the
African American population had a specific TTR mutation that significantly increases the
risk of TTR amyloidosis [7], while approximately eight percent of patients with suspected
cardiac amyloidosis having pathogenic TTR mutations [8].
Int. J. Mol. Sci. 2021, 22, 3488. https://doi.org/10.3390/ijms22073488 https://www.mdpi.com/journal/ijms