Assessing the consistency of iPSC and animal models in cystic fibrosis modelling: A meta-analysis
<h3>Introduction</h3><p dir="ltr">Cystic fibrosis (CF) is a hereditary autosomal recessive disorder caused by a range of mutations in the CF Transmembrane Conductance Regulator (CFTR) gene. This gene encodes the CFTR protein, which acts as a chloride channel activated by...
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| مؤلفون آخرون: | , , , , , |
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2022
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| _version_ | 1864513526471589888 |
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| author | Toqa Darwish (13222230) |
| author2 | Azhar Al-Khulaifi (13222233) Menatalla Ali (13222236) Rana Mowafy (13222239) Abdelilah Arredouani (10914455) Suhail A. Doi (9552474) Mohamed M. Emara (9913215) |
| author2_role | author author author author author author |
| author_facet | Toqa Darwish (13222230) Azhar Al-Khulaifi (13222233) Menatalla Ali (13222236) Rana Mowafy (13222239) Abdelilah Arredouani (10914455) Suhail A. Doi (9552474) Mohamed M. Emara (9913215) |
| author_role | author |
| dc.creator.none.fl_str_mv | Toqa Darwish (13222230) Azhar Al-Khulaifi (13222233) Menatalla Ali (13222236) Rana Mowafy (13222239) Abdelilah Arredouani (10914455) Suhail A. Doi (9552474) Mohamed M. Emara (9913215) |
| dc.date.none.fl_str_mv | 2022-08-09T03:00:00Z |
| dc.identifier.none.fl_str_mv | 10.1371/journal.pone.0272091 |
| dc.relation.none.fl_str_mv | https://figshare.com/articles/journal_contribution/Assessing_the_consistency_of_iPSC_and_animal_models_in_cystic_fibrosis_modelling_A_meta-analysis/25257238 |
| dc.rights.none.fl_str_mv | CC BY 4.0 info:eu-repo/semantics/openAccess |
| dc.subject.none.fl_str_mv | Biological sciences Genetics Zoology Biomedical and clinical sciences Cardiovascular medicine and haematology Medical biochemistry and metabolomics Medical biotechnology Mathematical sciences Statistics Cystic Fibrosis (CF) Autosomal recessive disorder CFTR gene CFTR protein Respiratory disease Pathogens Respiratory failure Liver, pancreas, small bowel Molecular mechanisms Disease modeling Animal models iPSC (Induced Pluripotent Stem Cells) Amyotrophic lateral sclerosis (ALS) Spinal muscular atrophy (SMA) Parkinson’s disease (PD) Alzheimer’s disease (AD) Drug responses Chloride channel function |
| dc.title.none.fl_str_mv | Assessing the consistency of iPSC and animal models in cystic fibrosis modelling: A meta-analysis |
| dc.type.none.fl_str_mv | Text Journal contribution info:eu-repo/semantics/publishedVersion text contribution to journal |
| description | <h3>Introduction</h3><p dir="ltr">Cystic fibrosis (CF) is a hereditary autosomal recessive disorder caused by a range of mutations in the CF Transmembrane Conductance Regulator (CFTR) gene. This gene encodes the CFTR protein, which acts as a chloride channel activated by cyclic AMP (cAMP). This meta-analysis aimed to compare the responsiveness of induced pluripotent stem cells (iPSCs) to cAMP analogues to that of commonly used animal models.</p><h3>Methods</h3><p dir="ltr">Databases searched included PubMed, Scopus, and Medline from inception to January 2020. A total of 8 and 3 studies, respectively, for animal models and iPSCs, were analyzed. Studies were extracted for investigating cAMP-stimulated anion transport by measuring the short circuit current (Isc) of chloride channels in different animal models and iPSC systems We utilized an inverse variance heterogeneity model for synthesis.</p><h3>Results</h3><p dir="ltr">Our analysis showed considerable heterogeneity in the mean Isc value in both animal models and iPSCs studies (compared to their WT counterparts), and both suffer from variable responsiveness based on the nature of the underlying model. There was no clear advantage of one over the other.</p><h3>Conclusions</h3><p dir="ltr">Studies on both animal and iPSCs models generated considerable heterogeneity. Given the potential of iPSC-derived models to study different diseases, we recommend paying more attention to developing reproducible models of iPSC as it has potential if adequately developed.</p><h2>Other Information</h2><p dir="ltr">Published in: PLOS ONE<br>License: <a href="http://creativecommons.org/licenses/by/4.0/" target="_blank">http://creativecommons.org/licenses/by/4.0/</a><br>See article on publisher's website: <a href="https://dx.doi.org/10.1371/journal.pone.0272091" target="_blank">https://dx.doi.org/10.1371/journal.pone.0272091</a></p> |
| eu_rights_str_mv | openAccess |
| id | Manara2_285a6793b9f1898b7ec48175d66c1b01 |
| identifier_str_mv | 10.1371/journal.pone.0272091 |
| network_acronym_str | Manara2 |
| network_name_str | Manara2 |
| oai_identifier_str | oai:figshare.com:article/25257238 |
| publishDate | 2022 |
| repository.mail.fl_str_mv | |
| repository.name.fl_str_mv | |
| repository_id_str | |
| rights_invalid_str_mv | CC BY 4.0 |
| spelling | Assessing the consistency of iPSC and animal models in cystic fibrosis modelling: A meta-analysisToqa Darwish (13222230)Azhar Al-Khulaifi (13222233)Menatalla Ali (13222236)Rana Mowafy (13222239)Abdelilah Arredouani (10914455)Suhail A. Doi (9552474)Mohamed M. Emara (9913215)Biological sciencesGeneticsZoologyBiomedical and clinical sciencesCardiovascular medicine and haematologyMedical biochemistry and metabolomicsMedical biotechnologyMathematical sciencesStatisticsCystic Fibrosis (CF)Autosomal recessive disorderCFTR geneCFTR proteinRespiratory diseasePathogensRespiratory failureLiver, pancreas, small bowelMolecular mechanismsDisease modelingAnimal modelsiPSC (Induced Pluripotent Stem Cells)Amyotrophic lateral sclerosis (ALS)Spinal muscular atrophy (SMA)Parkinson’s disease (PD)Alzheimer’s disease (AD)Drug responsesChloride channel function<h3>Introduction</h3><p dir="ltr">Cystic fibrosis (CF) is a hereditary autosomal recessive disorder caused by a range of mutations in the CF Transmembrane Conductance Regulator (CFTR) gene. This gene encodes the CFTR protein, which acts as a chloride channel activated by cyclic AMP (cAMP). This meta-analysis aimed to compare the responsiveness of induced pluripotent stem cells (iPSCs) to cAMP analogues to that of commonly used animal models.</p><h3>Methods</h3><p dir="ltr">Databases searched included PubMed, Scopus, and Medline from inception to January 2020. A total of 8 and 3 studies, respectively, for animal models and iPSCs, were analyzed. Studies were extracted for investigating cAMP-stimulated anion transport by measuring the short circuit current (Isc) of chloride channels in different animal models and iPSC systems We utilized an inverse variance heterogeneity model for synthesis.</p><h3>Results</h3><p dir="ltr">Our analysis showed considerable heterogeneity in the mean Isc value in both animal models and iPSCs studies (compared to their WT counterparts), and both suffer from variable responsiveness based on the nature of the underlying model. There was no clear advantage of one over the other.</p><h3>Conclusions</h3><p dir="ltr">Studies on both animal and iPSCs models generated considerable heterogeneity. Given the potential of iPSC-derived models to study different diseases, we recommend paying more attention to developing reproducible models of iPSC as it has potential if adequately developed.</p><h2>Other Information</h2><p dir="ltr">Published in: PLOS ONE<br>License: <a href="http://creativecommons.org/licenses/by/4.0/" target="_blank">http://creativecommons.org/licenses/by/4.0/</a><br>See article on publisher's website: <a href="https://dx.doi.org/10.1371/journal.pone.0272091" target="_blank">https://dx.doi.org/10.1371/journal.pone.0272091</a></p>2022-08-09T03:00:00ZTextJournal contributioninfo:eu-repo/semantics/publishedVersiontextcontribution to journal10.1371/journal.pone.0272091https://figshare.com/articles/journal_contribution/Assessing_the_consistency_of_iPSC_and_animal_models_in_cystic_fibrosis_modelling_A_meta-analysis/25257238CC BY 4.0info:eu-repo/semantics/openAccessoai:figshare.com:article/252572382022-08-09T03:00:00Z |
| spellingShingle | Assessing the consistency of iPSC and animal models in cystic fibrosis modelling: A meta-analysis Toqa Darwish (13222230) Biological sciences Genetics Zoology Biomedical and clinical sciences Cardiovascular medicine and haematology Medical biochemistry and metabolomics Medical biotechnology Mathematical sciences Statistics Cystic Fibrosis (CF) Autosomal recessive disorder CFTR gene CFTR protein Respiratory disease Pathogens Respiratory failure Liver, pancreas, small bowel Molecular mechanisms Disease modeling Animal models iPSC (Induced Pluripotent Stem Cells) Amyotrophic lateral sclerosis (ALS) Spinal muscular atrophy (SMA) Parkinson’s disease (PD) Alzheimer’s disease (AD) Drug responses Chloride channel function |
| status_str | publishedVersion |
| title | Assessing the consistency of iPSC and animal models in cystic fibrosis modelling: A meta-analysis |
| title_full | Assessing the consistency of iPSC and animal models in cystic fibrosis modelling: A meta-analysis |
| title_fullStr | Assessing the consistency of iPSC and animal models in cystic fibrosis modelling: A meta-analysis |
| title_full_unstemmed | Assessing the consistency of iPSC and animal models in cystic fibrosis modelling: A meta-analysis |
| title_short | Assessing the consistency of iPSC and animal models in cystic fibrosis modelling: A meta-analysis |
| title_sort | Assessing the consistency of iPSC and animal models in cystic fibrosis modelling: A meta-analysis |
| topic | Biological sciences Genetics Zoology Biomedical and clinical sciences Cardiovascular medicine and haematology Medical biochemistry and metabolomics Medical biotechnology Mathematical sciences Statistics Cystic Fibrosis (CF) Autosomal recessive disorder CFTR gene CFTR protein Respiratory disease Pathogens Respiratory failure Liver, pancreas, small bowel Molecular mechanisms Disease modeling Animal models iPSC (Induced Pluripotent Stem Cells) Amyotrophic lateral sclerosis (ALS) Spinal muscular atrophy (SMA) Parkinson’s disease (PD) Alzheimer’s disease (AD) Drug responses Chloride channel function |