Abstract:
:The zebrafish has been the model of choice amongst developmental biologists for many years. This small freshwater species offers many advantages to the study of organ and tissue development that are not provided by other model systems. Against this background, modern molecular genetic approaches are being applied to expand the physical and genetic mapping of the zebrafish genome. These approaches complement the large-scale mutagenic screens that have led to the isolation of mutant phenotypes. Some of the phenotypes have been found to resemble human disease states, while mapping and sequencing data have revealed zebrafish genes with significant homology to human disease-causing genes. It is the realization that the zebrafish offers an amenable system for understanding disease, as opposed to development, that underpins this review. The adventitious identification of disease phenotypes amongst zebrafish mutants and the important area of deliberate disease modelling using transgenesis and gene targeting should lead to a better application of the zebrafish as a vertebrate model of human diseases.
journal_name
Hum Mol Genetjournal_title
Human molecular geneticsauthors
Dodd A,Curtis PM,Williams LC,Love DRdoi
10.1093/hmg/9.16.2443subject
Has Abstractpub_date
2000-10-01 00:00:00pages
2443-9issue
16eissn
0964-6906issn
1460-2083journal_volume
9pub_type
杂志文章,评审abstract::Oculopharyngeal muscular dystrophy (OPMD) is a late-onset autosomal dominant muscular dystrophy that results from small expansions of a polyalanine tract in the PABPN1 gene. Intranuclear inclusions are the pathological hallmark of OPMD. The mechanism by which protein aggregation in OPMD might relate to a toxic gain-of...
journal_title:Human molecular genetics
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journal_title:Human molecular genetics
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journal_title:Human molecular genetics
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journal_title:Human molecular genetics
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更新日期:2014-02-15 00:00:00
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journal_title:Human molecular genetics
pub_type: 杂志文章
doi:10.1093/hmg/5.11.1733
更新日期:1996-11-01 00:00:00
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journal_title:Human molecular genetics
pub_type: 杂志文章
doi:10.1093/hmg/ddw204
更新日期:2016-08-15 00:00:00
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journal_title:Human molecular genetics
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doi:10.1093/hmg/11.15.1785
更新日期:2002-07-15 00:00:00
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journal_title:Human molecular genetics
pub_type: 杂志文章
doi:10.1093/hmg/ddz212
更新日期:2019-11-15 00:00:00
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journal_title:Human molecular genetics
pub_type: 杂志文章
doi:10.1093/hmg/11.16.1879
更新日期:2002-08-01 00:00:00
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journal_title:Human molecular genetics
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更新日期:2019-09-15 00:00:00
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journal_title:Human molecular genetics
pub_type: 杂志文章,评审
doi:10.1093/hmg/10.20.2187
更新日期:2001-10-01 00:00:00
abstract::A vast portion of intellectual disability and autism spectrum disorders is genetically caused by mutations in chromatin modulators. These proteins play key roles in development and are also highly expressed in the adult brain. Specifically, the pivotal role of chromatin regulation in transcription has placed enhancers...
journal_title:Human molecular genetics
pub_type: 杂志文章,评审
doi:10.1093/hmg/ddz196
更新日期:2019-11-21 00:00:00
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journal_title:Human molecular genetics
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更新日期:2011-12-01 00:00:00
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更新日期:2011-08-15 00:00:00
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pub_type: 杂志文章
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更新日期:2007-11-01 00:00:00
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journal_title:Human molecular genetics
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更新日期:1995-01-01 00:00:00
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journal_title:Human molecular genetics
pub_type: 杂志文章,评审
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journal_title:Human molecular genetics
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更新日期:2019-12-01 00:00:00
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journal_title:Human molecular genetics
pub_type: 杂志文章
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更新日期:2005-11-15 00:00:00
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journal_title:Human molecular genetics
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更新日期:2013-02-15 00:00:00