کد مقاله کد نشریه سال انتشار مقاله انگلیسی نسخه تمام متن
1931425 1050552 2010 6 صفحه PDF دانلود رایگان
عنوان انگلیسی مقاله ISI
Foxn3 is essential for craniofacial development in mice and a putative candidate involved in human congenital craniofacial defects
موضوعات مرتبط
علوم زیستی و بیوفناوری بیوشیمی، ژنتیک و زیست شناسی مولکولی زیست شیمی
پیش نمایش صفحه اول مقاله
Foxn3 is essential for craniofacial development in mice and a putative candidate involved in human congenital craniofacial defects
چکیده انگلیسی

The fork-head transcription factors are involved in a variety of physiological processes including development, aging, obesity, and cancer. The fork-head transcription factor FOXN3 has been implicated in cell cycle and transcription regulation at the cellular level. However, the physiological functions of FOXN3 in mammals are not known. To understand the role of the fork-head transcription factor FOXN3 in mammalian development, we have generated a mutant mouse model for the Foxn3 gene. Our analysis shows that the Foxn3 mutation leads to partial embryonic and postnatal lethality, growth retardation, eye formation defects, dental anomalies and craniofacial defects. Foxn3 mutant tissues and cells are also defective in the expression of distinct osteogenic genes. Interestingly, the phenotypes of Foxn3 mutant mice show a striking overlap with the clinical features of human patients with congenital defects and chromosomal aberrations involving the human FOXN3 locus. More than three fourths of human congenital disorders involve craniofacial malformations and a majority of the perturbed genetic components that lead to such disorders are yet to be identified. Our results implicate a role for the FOXN3 gene in the etiology of craniofacial defects in humans.

Research highlights
► A novel gene-trap mutation unravels the role of Foxn3 in mammalian development.
► Foxn3 affects craniofacial development in mice.
► Foxn3 regulates the expression of osteogenic genes during craniofacial development.
► Mutant mice phenotypes and clinical features of human congenital patients overlap.

ناشر
Database: Elsevier - ScienceDirect (ساینس دایرکت)
Journal: Biochemical and Biophysical Research Communications - Volume 400, Issue 1, 10 September 2010, Pages 60–65
نویسندگان
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