کد مقاله کد نشریه سال انتشار مقاله انگلیسی نسخه تمام متن
5531893 1401819 2017 13 صفحه PDF دانلود رایگان
عنوان انگلیسی مقاله ISI
Original research articleZebrafish zic2 controls formation of periocular neural crest and choroid fissure morphogenesis
موضوعات مرتبط
علوم زیستی و بیوفناوری بیوشیمی، ژنتیک و زیست شناسی مولکولی بیولوژی سلول
پیش نمایش صفحه اول مقاله
Original research articleZebrafish zic2 controls formation of periocular neural crest and choroid fissure morphogenesis
چکیده انگلیسی


- Zic2 controls choroid fissure morphogenesis in zebrafish.
- Zic2 is required for periocular mesenchyme formation.
- Zic2 activates transcription of alx1, a transcription factor with essential functions during craniofacial and retinal development.

The vertebrate retina develops in close proximity to the forebrain and neural crest-derived cartilages of the face and jaw. Coloboma, a congenital eye malformation, is associated with aberrant forebrain development (holoprosencephaly) and with craniofacial defects (frontonasal dysplasia) in humans, suggesting a critical role for cross-lineage interactions during retinal morphogenesis. ZIC2, a zinc-finger transcription factor, is linked to human holoprosencephaly. We have previously used morpholino assays to show zebrafish zic2 functions in the developing forebrain, retina and craniofacial cartilage. We now report that zebrafish with genetic lesions in zebrafish zic2 orthologs, zic2a and zic2b, develop with retinal coloboma and craniofacial anomalies. We demonstrate a requirement for zic2 in restricting pax2a expression and show evidence that zic2 function limits Hh signaling. RNA-seq transcriptome analysis identified an early requirement for zic2 in periocular neural crest as an activator of alx1, a transcription factor with essential roles in craniofacial and ocular morphogenesis in human and zebrafish. Collectively, these data establish zic2 mutant zebrafish as a powerful new genetic model for in-depth dissection of cell interactions and genetic controls during craniofacial complex development.

ناشر
Database: Elsevier - ScienceDirect (ساینس دایرکت)
Journal: Developmental Biology - Volume 429, Issue 1, 1 September 2017, Pages 92-104
نویسندگان
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