Passa al contenuto
Merck

Enzyme enhancers for the treatment of Fabry and Pompe disease.

Molecular therapy : the journal of the American Society of Gene Therapy (2014-11-21)
Jan Lukas, Anne-Marie Pockrandt, Susanne Seemann, Muhammad Sharif, Franziska Runge, Susann Pohlers, Chaonan Zheng, Anne Gläser, Matthias Beller, Arndt Rolfs, Anne-Katrin Giese
ABSTRACT

Lysosomal storage disorders (LSD) are a group of heterogeneous diseases caused by compromised enzyme function leading to multiple organ failure. Therapeutic approaches involve enzyme replacement (ERT), which is effective for a substantial fraction of patients. However, there are still concerns about a number of issues including tissue penetrance, generation of host antibodies against the therapeutic enzyme, and financial aspects, which render this therapy suboptimal for many cases. Treatment with pharmacological chaperones (PC) was recognized as a possible alternative to ERT, because a great number of mutations do not completely abolish enzyme function, but rather trigger degradation in the endoplasmic reticulum. The theory behind PC is that they can stabilize enzymes with remaining function, avoid degradation and thereby ameliorate disease symptoms. We tested several compounds in order to identify novel small molecules that prevent premature degradation of the mutant lysosomal enzymes α-galactosidase A (for Fabry disease (FD)) and acid α-glucosidase (GAA) (for Pompe disease (PD)). We discovered that the expectorant Ambroxol when used in conjunction with known PC resulted in a significant enhancement of mutant α-galactosidase A and GAA activities. Rosiglitazone was effective on α-galactosidase A either as a monotherapy or when administered in combination with the PC 1-deoxygalactonojirimycin. We therefore propose both drugs as potential enhancers of pharmacological chaperones in FD and PD to improve current treatment strategies.

MATERIALI
N° Catalogo
Marchio
Descrizione del prodotto

Sigma-Aldrich
Acido cloridrico, ACS reagent, 37%
Sigma-Aldrich
Acido cloridrico, ACS reagent, 37%
Sigma-Aldrich
Cloruro di idrogeno, 4.0 M in dioxane
Sigma-Aldrich
Acido cloridrico, 1.0 N, BioReagent, suitable for cell culture
Sigma-Aldrich
Acido cloridrico, 37 wt. % in H2O, 99.999% trace metals basis
Sigma-Aldrich
Acido cloridrico, puriss. p.a., ACS reagent, reag. ISO, reag. Ph. Eur., fuming, ≥37%, APHA: ≤10
Sigma-Aldrich
Acido cloridrico, meets analytical specification of Ph. Eur., BP, NF, fuming, 36.5-38%
Sigma-Aldrich
Acido cloridrico, 36.5-38.0%, BioReagent, for molecular biology
Sigma-Aldrich
Cloruro di idrogeno, 2.0 M in diethyl ether
Sigma-Aldrich
D-(+)-Galattosio, ≥99% (HPLC), BioReagent, suitable for cell culture, suitable for insect cell culture
Sigma-Aldrich
Tapsigargina, ≥98% (HPLC), solid film
Supelco
Acido cloridrico, volumetric, 0.1 M HCl (0.1N), endotoxin free
Sigma-Aldrich
Cloruro di idrogeno, 1.0 M in diethyl ether
Sigma-Aldrich
Hydrogen chloride, ReagentPlus®, ≥99%
Sigma-Aldrich
Acido cloridrico, puriss., 24.5-26.0%
Sigma-Aldrich
D-(+)-Galattosio, ≥99% (HPLC)
Sigma-Aldrich
Acido cloridrico, ~6 M in H2O, for amino acid analysis
Sigma-Aldrich
Cloruro di idrogeno, 3 M in cyclopentyl methyl ether (CPME)
Sigma-Aldrich
Acido cloridrico, 32 wt. % in H2O, FCC
Sigma-Aldrich
D-(+)-Galattosio, ≥98% (HPLC)
Sigma-Aldrich
Cloruro di idrogeno, 1.0 M in acetic acid
Supelco
D-(+)-Galattosio, Pharmaceutical Secondary Standard; Certified Reference Material
USP
Galattosio, United States Pharmacopeia (USP) Reference Standard
Supelco
Hydrogen chloride – methanol solution, ~1.25 m HCl (T), for GC derivatization, LiChropur
Sigma-Aldrich
Bezafibrate, ≥98%, solid
Sigma-Aldrich
Lactacystin, ≥90% (HPLC)
Supelco
Hydrogen chloride – ethanol solution, ~1.25 M HCl, for GC derivatization, LiChropur
Millipore
D-(+)-Galattosio, suitable for microbiology, ≥99.0%
Sigma-Aldrich
Acido bicinconinico, ≥98% (HPLC)
Ambroxolo, European Pharmacopoeia (EP) Reference Standard