Long-term evolution of mucopolysaccharidosis type I in twins treated with enzyme replacement therapy plus hematopoietic stem cells transplantation.

Heliyon

Bone Marrow Transplantation Unit, Instituto Nacional de Pediatría, Av Insurgentes Sur 3700 c Insurgentes Cuicuilco, Mexico City, 04530, Mexico.

Published: August 2021

Mucopolysaccharidoses (MPSs) are a heterogeneous group of diseases that have in common the accumulation of glycosaminoglycans (mucopolysaccharides) within the lysosome. The diseases are caused by a deficiency of the enzyme α-L-iduronidase which is responsible for the degradation of glycosaminoglycans (GAGs or mucopolysaccharides). More than 100 mutations in the gene have been reported, resulting in marked clinical/response variability. MPSs usually present as multisystem and progressive clinical disorders which affect psychomotor and cardiovascular development, the cornea and the musculoskeletal system. Seven phenotypically distinct diseases have been described, and MPS type I (MPS-I) is divided into three clinical forms: severe (Hurler syndrome), intermediate (Hurler-Scheie syndrome) or mild (Scheie syndrome). For the treatment of MPS-I, Enzyme Replacement Therapy (ERT) with α-L-iduronidase and Hematopoietic Stem Cells Transplantation (HSCT), separately or in combination, have produced clinical improvement, especially with regards cardiovascular symptoms and psychomotor development. This article presents the long-term (more than seven years) follow-up of monochorionic, diamniotic twins who were diagnosed with MPS-I at an early stage, and treated with ERT (from age 10 months) plus HSCT (from age 18 months). Overall, the treatment has facilitated stable development with an overall good response and better control of symptoms associated with MPS-I.

Download full-text PDF

Source
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8379671PMC
http://dx.doi.org/10.1016/j.heliyon.2021.e07740DOI Listing

Publication Analysis

Top Keywords

enzyme replacement
8
replacement therapy
8
hematopoietic stem
8
stem cells
8
cells transplantation
8
age months
8
long-term evolution
4
evolution mucopolysaccharidosis
4
mucopolysaccharidosis type
4
type twins
4

Similar Publications

Triple-negative breast cancer (TNBC) is one of the most fatal malignancies in the world, accounting for 42% of all deaths due to metastasis. The significant development is hindered by the multi-drug resistance and poor patient compliance. PIK3CA gene mutation is one of the important causes of TNBC, which causes dysregulation of the cell cycle and cell proliferation.

View Article and Find Full Text PDF

Objective: The study aimed to evaluate the epidemiological, clinical, and molecular data of mucopolysaccharidosis type II (MPS II) patients and their outcomes using the national registry of patients in the Russian Federation (RF). Materials and Methods: In the retrospective cohort study, the authors included data from the Russian national registry of MPS II. Results: The prevalence of MPS II in RF is 0.

View Article and Find Full Text PDF

Background: The current standard of care for infantile-onset Pompe disease (IOPD), a severe form of acid α-glucosidase enzyme activity deficiency is: (1) detection by newborn screening, (2) early initiation of intravenous enzyme replacement therapy (ERT) using recombinant human acid alpha-glucosidase (rhGAA), with higher doses of rhGAA increasingly used to improve clinical outcomes, and (3) immune tolerization induction (ITI) using to prevent anti-rhGAA antibody formation, with methotrexate (MTX), rituximab, and IVIG used for patients who are cross-reactive immunologic material negative (CRIM-) and monotherapy with MTX used in patients who are cross-reactive immunologic material positive (CRIM+).

Objectives/methods: A pilot study evaluates a dose-intensive therapy (DIT) using high-dose ERT (40 mg/kg/week) and more frequent exposure to ERT (i.e.

View Article and Find Full Text PDF

A novel antimonotungstate (AT)-based heterometallic framework {[Er(HO)][Fe(Hpdc)(B-β-SbWO)]}·50HO (, Hpdc = pyridine-2,5-dicarboxylic acid) was obtained through a synergistic strategy of in situ-generated transition-metal-encapsulated polyoxometalate (POM) building units and the substitution reaction. Its structural unit is composed of a tetra-Fe-substituted Krebs-type [Fe(Hpdc)(B-β-SbWO)] subunit and two [Er(HO)] cations. This subunit can be regarded as a product of carboxylic oxygen atoms of Hpdc ligands replacing active water ligands in the [Fe(HO)(B-β-SbWO)] species.

View Article and Find Full Text PDF

Want AI Summaries of new PubMed Abstracts delivered to your In-box?

Enter search terms and have AI summaries delivered each week - change queries or unsubscribe any time!