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Osteogenesis Imperfecta: Current and Prospective Therapies

Osteogenesis Imperfecta (OI) is a group of connective tissue disorders with a broad range of phenotypes characterized primarily by bone fragility. The prevalence of OI ranges from about 1:15,000 to 1:20,000 births. Five types of the disease are commonly distinguished, ranging from a mild (type I) to...

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Autores principales: Botor, Malwina, Fus-Kujawa, Agnieszka, Uroczynska, Marta, Stepien, Karolina L., Galicka, Anna, Gawron, Katarzyna, Sieron, Aleksander L.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8533546/
https://www.ncbi.nlm.nih.gov/pubmed/34680126
http://dx.doi.org/10.3390/biom11101493
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author Botor, Malwina
Fus-Kujawa, Agnieszka
Uroczynska, Marta
Stepien, Karolina L.
Galicka, Anna
Gawron, Katarzyna
Sieron, Aleksander L.
author_facet Botor, Malwina
Fus-Kujawa, Agnieszka
Uroczynska, Marta
Stepien, Karolina L.
Galicka, Anna
Gawron, Katarzyna
Sieron, Aleksander L.
author_sort Botor, Malwina
collection PubMed
description Osteogenesis Imperfecta (OI) is a group of connective tissue disorders with a broad range of phenotypes characterized primarily by bone fragility. The prevalence of OI ranges from about 1:15,000 to 1:20,000 births. Five types of the disease are commonly distinguished, ranging from a mild (type I) to a lethal one (type II). Types III and IV are severe forms allowing survival after the neonatal period, while type V is characterized by a mild to moderate phenotype with calcification of interosseous membranes. In most cases, there is a reduction in the production of normal type I collagen (col I) or the synthesis of abnormal collagen as a result of mutations in col I genes. Moreover, mutations in genes involved in col I synthesis and processing as well as in osteoblast differentiation have been reported. The currently available treatments try to prevent fractures, control symptoms and increase bone mass. Commonly used medications in OI treatment are bisphosphonates, Denosumab, synthetic parathyroid hormone and growth hormone for children therapy. The main disadvantages of these therapies are their relatively weak effectiveness, lack of effects in some patients or cytotoxic side effects. Experimental approaches, particularly those based on stem cell transplantation and genetic engineering, seem to be promising to improve the therapeutic effects of OI.
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spelling pubmed-85335462021-10-23 Osteogenesis Imperfecta: Current and Prospective Therapies Botor, Malwina Fus-Kujawa, Agnieszka Uroczynska, Marta Stepien, Karolina L. Galicka, Anna Gawron, Katarzyna Sieron, Aleksander L. Biomolecules Review Osteogenesis Imperfecta (OI) is a group of connective tissue disorders with a broad range of phenotypes characterized primarily by bone fragility. The prevalence of OI ranges from about 1:15,000 to 1:20,000 births. Five types of the disease are commonly distinguished, ranging from a mild (type I) to a lethal one (type II). Types III and IV are severe forms allowing survival after the neonatal period, while type V is characterized by a mild to moderate phenotype with calcification of interosseous membranes. In most cases, there is a reduction in the production of normal type I collagen (col I) or the synthesis of abnormal collagen as a result of mutations in col I genes. Moreover, mutations in genes involved in col I synthesis and processing as well as in osteoblast differentiation have been reported. The currently available treatments try to prevent fractures, control symptoms and increase bone mass. Commonly used medications in OI treatment are bisphosphonates, Denosumab, synthetic parathyroid hormone and growth hormone for children therapy. The main disadvantages of these therapies are their relatively weak effectiveness, lack of effects in some patients or cytotoxic side effects. Experimental approaches, particularly those based on stem cell transplantation and genetic engineering, seem to be promising to improve the therapeutic effects of OI. MDPI 2021-10-10 /pmc/articles/PMC8533546/ /pubmed/34680126 http://dx.doi.org/10.3390/biom11101493 Text en © 2021 by the authors. https://creativecommons.org/licenses/by/4.0/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/).
spellingShingle Review
Botor, Malwina
Fus-Kujawa, Agnieszka
Uroczynska, Marta
Stepien, Karolina L.
Galicka, Anna
Gawron, Katarzyna
Sieron, Aleksander L.
Osteogenesis Imperfecta: Current and Prospective Therapies
title Osteogenesis Imperfecta: Current and Prospective Therapies
title_full Osteogenesis Imperfecta: Current and Prospective Therapies
title_fullStr Osteogenesis Imperfecta: Current and Prospective Therapies
title_full_unstemmed Osteogenesis Imperfecta: Current and Prospective Therapies
title_short Osteogenesis Imperfecta: Current and Prospective Therapies
title_sort osteogenesis imperfecta: current and prospective therapies
topic Review
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8533546/
https://www.ncbi.nlm.nih.gov/pubmed/34680126
http://dx.doi.org/10.3390/biom11101493
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