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Generation of a new six1-null line in Xenopus tropicalis for study of development and congenital disease.
- Source :
-
Genesis (New York, N.Y. : 2000) [Genesis] 2021 Dec; Vol. 59 (12), pp. e23453. Date of Electronic Publication: 2021 Oct 19. - Publication Year :
- 2021
-
Abstract
- The vertebrate Six (Sine oculis homeobox) family of homeodomain transcription factors plays critical roles in the development of several organs. Six1 plays a central role in cranial placode development, including the precursor tissues of the inner ear, as well as other cranial sensory organs and the kidney. In humans, mutations in SIX1 underlie some cases of Branchio-oto-renal (BOR) syndrome, which is characterized by moderate-to-severe hearing loss. We utilized CRISPR/Cas9 technology to establish a six1 mutant line in Xenopus tropicalis that is available to the research community. We demonstrate that at larval stages, the six1-null animals show severe disruptions in gene expression of putative Six1 target genes in the otic vesicle, cranial ganglia, branchial arch, and neural tube. At tadpole stages, six1-null animals display dysmorphic Meckel's, ceratohyal, and otic capsule cartilage morphology. This mutant line will be of value for the study of the development of several organs as well as congenital syndromes that involve these tissues.<br /> (© 2021 Wiley Periodicals LLC.)
- Subjects :
- Animals
Branchial Region growth & development
Branchial Region pathology
Branchio-Oto-Renal Syndrome physiopathology
CRISPR-Cas Systems genetics
Congenital Abnormalities pathology
Embryonic Development genetics
Ganglia, Parasympathetic growth & development
Ganglia, Parasympathetic pathology
Gene Expression
Gene Expression Regulation, Developmental genetics
Hearing Loss physiopathology
Humans
Neural Tube growth & development
Neural Tube pathology
Skull growth & development
Skull pathology
Transcription Factors genetics
Xenopus genetics
Xenopus growth & development
Branchio-Oto-Renal Syndrome genetics
Congenital Abnormalities genetics
Hearing Loss genetics
Homeodomain Proteins genetics
Xenopus Proteins genetics
Subjects
Details
- Language :
- English
- ISSN :
- 1526-968X
- Volume :
- 59
- Issue :
- 12
- Database :
- MEDLINE
- Journal :
- Genesis (New York, N.Y. : 2000)
- Publication Type :
- Academic Journal
- Accession number :
- 34664392
- Full Text :
- https://doi.org/10.1002/dvg.23453