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Nosip functions during vertebrate eye and cranial cartilage development
Author(s) -
Flach Hannah,
Krieg Julia,
Hoffmeister Meike,
Dietmann Petra,
Reusch Adrian,
Wischmann Lisa,
Kernl Bianka,
Riegger Ricarda,
Oess Stefanie,
Kühl Susanne J.
Publication year - 2018
Publication title -
developmental dynamics
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 1.634
H-Index - 141
eISSN - 1097-0177
pISSN - 1058-8388
DOI - 10.1002/dvdy.24659
Subject(s) - xenopus , biology , neural crest , neurogenesis , pax6 , microbiology and biotechnology , neural development , neural plate , eye development , zebrafish , retina , retinoic acid , anatomy , retinal , cranial neural crest , neuroscience , embryo , genetics , transcription factor , gene , biochemistry
Background: The nitric oxide synthase interacting protein (Nosip) has been associated with diverse human diseases including psychological disorders. In line, early neurogenesis of mouse and Xenopus is impaired upon Nosip deficiency. Nosip knockout mice show craniofacial defects and the down‐regulation of Nosip in the mouse and Xenopus leads to microcephaly. Until now, the exact underlying molecular mechanisms of these malformations were still unknown. Results: Here, we show that nosip is expressed in the developing ocular system as well as the anterior neural crest cells of Xenopus laevis . Furthermore, Nosip inhibition causes severe defects in eye formation in the mouse and Xenopus . Retinal lamination as well as dorso–ventral patterning of the retina were affected in Nosip‐depleted Xenopus embryos. Marker gene analysis using rax, pax6 and otx2 reveals an interference with the eye field induction and differentiation. A closer look on Nosip‐deficient Xenopus embryos furthermore reveals disrupted cranial cartilage structures and an inhibition of anterior neural crest cell induction and migration shown by twist, snai2 , and egr2 . Moreover, foxc1 as downstream factor of retinoic acid signalling is affected upon Nosip deficiency. Conclusions: Nosip is a crucial factor for the development of anterior neural tissue such the eyes and neural crest cells. Developmental Dynamics 247:1070‐1082, 2018 . © 2018 Wiley Periodicals, Inc.