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Organoid Technology and Its Role for Theratyping Applications in Cystic Fibrosis

Cystic fibrosis (CF) is a autosomal recessive, multisystemic disease caused by different mutations in the CFTR gene encoding CF transmembrane conductance regulator. Although symptom management is important to avoid complications, the approval of CFTR modulator drugs in the clinic has demonstrated si...

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Detalles Bibliográficos
Autores principales: Conti, Jessica, Sorio, Claudio, Melotti, Paola
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9856584/
https://www.ncbi.nlm.nih.gov/pubmed/36670555
http://dx.doi.org/10.3390/children10010004
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author Conti, Jessica
Sorio, Claudio
Melotti, Paola
author_facet Conti, Jessica
Sorio, Claudio
Melotti, Paola
author_sort Conti, Jessica
collection PubMed
description Cystic fibrosis (CF) is a autosomal recessive, multisystemic disease caused by different mutations in the CFTR gene encoding CF transmembrane conductance regulator. Although symptom management is important to avoid complications, the approval of CFTR modulator drugs in the clinic has demonstrated significant improvements by targeting the primary molecular defect of CF and thereby preventing problems related to CFTR deficiency or dysfunction. CFTR modulator therapies have positively changed the patients’ quality of life, especially for those who start their use at the onset of the disease. Due to early diagnosis with the implementation of newborn screening programs and considerable progress in the treatment options, nowadays pediatric mortality was dramatically reduced. In any case, the main obstacle to treat CF is to predict the drug response of patients due to genetic complexity and heterogeneity. Advances in 3D culture systems have led to the extrapolation of disease modeling and individual drug response in vitro by producing mini organs called “organoids” easily obtained from nasal and rectal mucosa biopsies. In this review, we focus primarily on patient-derived intestinal organoids used as in vitro model for CF disease. Organoids combine high-validity of outcomes with a high throughput, thus enabling CF disease classification, drug development and treatment optimization in a personalized manner.
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spelling pubmed-98565842023-01-21 Organoid Technology and Its Role for Theratyping Applications in Cystic Fibrosis Conti, Jessica Sorio, Claudio Melotti, Paola Children (Basel) Review Cystic fibrosis (CF) is a autosomal recessive, multisystemic disease caused by different mutations in the CFTR gene encoding CF transmembrane conductance regulator. Although symptom management is important to avoid complications, the approval of CFTR modulator drugs in the clinic has demonstrated significant improvements by targeting the primary molecular defect of CF and thereby preventing problems related to CFTR deficiency or dysfunction. CFTR modulator therapies have positively changed the patients’ quality of life, especially for those who start their use at the onset of the disease. Due to early diagnosis with the implementation of newborn screening programs and considerable progress in the treatment options, nowadays pediatric mortality was dramatically reduced. In any case, the main obstacle to treat CF is to predict the drug response of patients due to genetic complexity and heterogeneity. Advances in 3D culture systems have led to the extrapolation of disease modeling and individual drug response in vitro by producing mini organs called “organoids” easily obtained from nasal and rectal mucosa biopsies. In this review, we focus primarily on patient-derived intestinal organoids used as in vitro model for CF disease. Organoids combine high-validity of outcomes with a high throughput, thus enabling CF disease classification, drug development and treatment optimization in a personalized manner. MDPI 2022-12-20 /pmc/articles/PMC9856584/ /pubmed/36670555 http://dx.doi.org/10.3390/children10010004 Text en © 2022 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
Conti, Jessica
Sorio, Claudio
Melotti, Paola
Organoid Technology and Its Role for Theratyping Applications in Cystic Fibrosis
title Organoid Technology and Its Role for Theratyping Applications in Cystic Fibrosis
title_full Organoid Technology and Its Role for Theratyping Applications in Cystic Fibrosis
title_fullStr Organoid Technology and Its Role for Theratyping Applications in Cystic Fibrosis
title_full_unstemmed Organoid Technology and Its Role for Theratyping Applications in Cystic Fibrosis
title_short Organoid Technology and Its Role for Theratyping Applications in Cystic Fibrosis
title_sort organoid technology and its role for theratyping applications in cystic fibrosis
topic Review
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9856584/
https://www.ncbi.nlm.nih.gov/pubmed/36670555
http://dx.doi.org/10.3390/children10010004
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