Modeling congenital disease and inborn errors of development in Drosophila melanogaster
Fly models that faithfully recapitulate various aspects of human disease and human health-related biology are being used for research into disease diagnosis and prevention. Established and new genetic strategies in Drosophila have yielded numerous substantial successes in modeling congenital disorde...
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| Format: | Online |
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The Company of Biologists Ltd
2016
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| Online Access: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4826979/ |
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pubmed-48269792016-05-19 Modeling congenital disease and inborn errors of development in Drosophila melanogaster Moulton, Matthew J. Letsou, Anthea Review Fly models that faithfully recapitulate various aspects of human disease and human health-related biology are being used for research into disease diagnosis and prevention. Established and new genetic strategies in Drosophila have yielded numerous substantial successes in modeling congenital disorders or inborn errors of human development, as well as neurodegenerative disease and cancer. Moreover, although our ability to generate sequence datasets continues to outpace our ability to analyze these datasets, the development of high-throughput analysis platforms in Drosophila has provided access through the bottleneck in the identification of disease gene candidates. In this Review, we describe both the traditional and newer methods that are facilitating the incorporation of Drosophila into the human disease discovery process, with a focus on the models that have enhanced our understanding of human developmental disorders and congenital disease. Enviable features of the Drosophila experimental system, which make it particularly useful in facilitating the much anticipated move from genotype to phenotype (understanding and predicting phenotypes directly from the primary DNA sequence), include its genetic tractability, the low cost for high-throughput discovery, and a genome and underlying biology that are highly evolutionarily conserved. In embracing the fly in the human disease-gene discovery process, we can expect to speed up and reduce the cost of this process, allowing experimental scales that are not feasible and/or would be too costly in higher eukaryotes. The Company of Biologists Ltd 2016-03-01 /pmc/articles/PMC4826979/ /pubmed/26935104 http://dx.doi.org/10.1242/dmm.023564 Text en © 2016. Published by The Company of Biologists Ltd http://creativecommons.org/licenses/by/3.0 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/3.0), which permits unrestricted use, distribution and reproduction in any medium provided that the original work is properly attributed. |
| repository_type |
Open Access Journal |
| institution_category |
Foreign Institution |
| institution |
US National Center for Biotechnology Information |
| building |
NCBI PubMed |
| collection |
Online Access |
| language |
English |
| format |
Online |
| author |
Moulton, Matthew J. Letsou, Anthea |
| spellingShingle |
Moulton, Matthew J. Letsou, Anthea Modeling congenital disease and inborn errors of development in Drosophila melanogaster |
| author_facet |
Moulton, Matthew J. Letsou, Anthea |
| author_sort |
Moulton, Matthew J. |
| title |
Modeling congenital disease and inborn errors of development in Drosophila melanogaster |
| title_short |
Modeling congenital disease and inborn errors of development in Drosophila melanogaster |
| title_full |
Modeling congenital disease and inborn errors of development in Drosophila melanogaster |
| title_fullStr |
Modeling congenital disease and inborn errors of development in Drosophila melanogaster |
| title_full_unstemmed |
Modeling congenital disease and inborn errors of development in Drosophila melanogaster |
| title_sort |
modeling congenital disease and inborn errors of development in drosophila melanogaster |
| description |
Fly models that faithfully recapitulate various aspects of human disease and human health-related biology are being used for research into disease diagnosis and prevention. Established and new genetic strategies in Drosophila have yielded numerous substantial successes in modeling congenital disorders or inborn errors of human development, as well as neurodegenerative disease and cancer. Moreover, although our ability to generate sequence datasets continues to outpace our ability to analyze these datasets, the development of high-throughput analysis platforms in Drosophila has provided access through the bottleneck in the identification of disease gene candidates. In this Review, we describe both the traditional and newer methods that are facilitating the incorporation of Drosophila into the human disease discovery process, with a focus on the models that have enhanced our understanding of human developmental disorders and congenital disease. Enviable features of the Drosophila experimental system, which make it particularly useful in facilitating the much anticipated move from genotype to phenotype (understanding and predicting phenotypes directly from the primary DNA sequence), include its genetic tractability, the low cost for high-throughput discovery, and a genome and underlying biology that are highly evolutionarily conserved. In embracing the fly in the human disease-gene discovery process, we can expect to speed up and reduce the cost of this process, allowing experimental scales that are not feasible and/or would be too costly in higher eukaryotes. |
| publisher |
The Company of Biologists Ltd |
| publishDate |
2016 |
| url |
https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4826979/ |
| _version_ |
1613564499391938560 |