TY - JOUR
T1 - A perspective on research, diagnosis, and management of lysosomal storage disorders in Colombia
AU - Puentes-Tellez, María Alejandra
AU - Lerma-Barbosa, Paula Andrea
AU - Garzón-Jaramillo, Rafael Guillermo
AU - Suarez, Diego A.
AU - Espejo-Mojica, Angela J.
AU - Guevara, Johana M.
AU - Echeverri, Olga Yaneth
AU - Solano-Galarza, Daniela
AU - Uribe-Ardila, Alfredo
AU - Alméciga-Díaz, Carlos J.
N1 - Publisher Copyright:
© 2020 The Author(s)
PY - 2020/3
Y1 - 2020/3
N2 - Lysosomal storage diseases (LSDs) are a group of about 50 inborn errors of metabolism characterized by the lysosomal accumulation of partially or non-degraded molecules due to mutations in proteins involved in the degradation of macromolecules, transport, lysosomal biogenesis or modulators of lysosomal environment. Significant advances have been achieved in the diagnosis, management, and treatment of LSDs patients. In terms of approved therapies, these include enzyme replacement therapy (ERT), substrate reduction therapy, hematopoietic stem cell transplantation, and pharmacological chaperone therapy. In this review, we summarize the Colombian experience in LSDs thorough the evidence published. We identified 113 articles published between 1995 and 2019 that included Colombian researchers or physicians, and which were mainly focused in Mucopolysaccharidoses, Pompe disease, Gaucher disease, Fabry disease, and Tay-Sachs and Sandhoff diseases. Most of these articles focused on basic research, clinical cases, and mutation reports. Noteworthy, implementation of the enzyme assay in dried blood samples, led to a 5-fold increase in the identification of LSD patients, suggesting that these disorders still remain undiagnosed in the country. We consider that the information presented in this review will contribute to the knowledge of a broad spectrum of LSDs in Colombia and will also contribute to the development of public policies and the identification of research opportunities.
AB - Lysosomal storage diseases (LSDs) are a group of about 50 inborn errors of metabolism characterized by the lysosomal accumulation of partially or non-degraded molecules due to mutations in proteins involved in the degradation of macromolecules, transport, lysosomal biogenesis or modulators of lysosomal environment. Significant advances have been achieved in the diagnosis, management, and treatment of LSDs patients. In terms of approved therapies, these include enzyme replacement therapy (ERT), substrate reduction therapy, hematopoietic stem cell transplantation, and pharmacological chaperone therapy. In this review, we summarize the Colombian experience in LSDs thorough the evidence published. We identified 113 articles published between 1995 and 2019 that included Colombian researchers or physicians, and which were mainly focused in Mucopolysaccharidoses, Pompe disease, Gaucher disease, Fabry disease, and Tay-Sachs and Sandhoff diseases. Most of these articles focused on basic research, clinical cases, and mutation reports. Noteworthy, implementation of the enzyme assay in dried blood samples, led to a 5-fold increase in the identification of LSD patients, suggesting that these disorders still remain undiagnosed in the country. We consider that the information presented in this review will contribute to the knowledge of a broad spectrum of LSDs in Colombia and will also contribute to the development of public policies and the identification of research opportunities.
KW - Biochemistry
KW - Clinical genetics
KW - Clinical research
KW - Colombia
KW - Epidemiology
KW - Fabry
KW - Gaucher
KW - Genetics
KW - Laboratory medicine
KW - Lysosomal storage diseases
KW - Metabolite
KW - Mucopolysaccharidoses
KW - Pharmaceutical science
UR - http://www.scopus.com/inward/record.url?scp=85082442877&partnerID=8YFLogxK
U2 - 10.1016/j.heliyon.2020.e03635
DO - 10.1016/j.heliyon.2020.e03635
M3 - Review article
AN - SCOPUS:85082442877
SN - 2405-8440
VL - 6
JO - Heliyon
JF - Heliyon
IS - 3
M1 - e03635
ER -