The mouse slalom mutant demonstrates a role for Jagged1 in neuroepithelial patterning in the organ of Corti

Tsai, Hsun; Hardisty, Rachel E.; Rhodes, Charlotte; Kiernan, Amy E.; Roby, Phil; Tymowska-Lalanne, Zuzanna; Mburu, Philomena; Rastan, Sohaila; Hunter, A. Jackie; Brown, Steve; Steel, Karen P.

doi:10.1093/hmg/10.5.507

Cited by 127 publications

(99 citation statements)

References 24 publications

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“…The ENU-induced Jag1 alleles slalom and headturner Tsai et al 2001;Vrijens et al 2006) were generated using two different C3H substrains (C3HeJ and C3HeB/FeJ). We investigated the effects of genetic background on the Jag1 del1 /1 mutant phenotype by crossing Jag1 del1 /1 mice on the C57BL/6J (B6) background (B6-Jag1 del1 /1) to C3HeB/FeJ (hereafter designated C3H) mice.…”

Section: Resultsmentioning

confidence: 99%

Genetic Background Modifies Inner Ear and Eye Phenotypes of Jag1 Heterozygous Mice

Kiernan¹,

Hawes

et al. 2007

Genetics

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Mice heterozygous for missense mutations of the Notch ligand Jagged1 ( Jag1) exhibit head-shaking behavior indicative of an inner ear vestibular defect. In contrast, mice heterozygous for a targeted deletion of the Jag1 gene ( Jag1 del1 ) do not demonstrate obvious head-shaking behavior. To determine whether the differences in inner ear phenotypes were due to the types of Jag1 mutations or to differences in genetic background, we crossed Jag1 del1 heterozygous mice onto the same genetic background as the missense mutants. This analysis revealed that variation of the Jag1 mutant inner ear phenotype is caused by genetic background differences and is not due to the type of Jag1 mutation. Genome scans of N2 backcross mice identified a significant modifier locus on chromosome 7, as well as a suggestive locus on chromosome 14. We also analyzed modifiers of an eye defect in Jag1 del1 heterozygous mice from this same cross.

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Section: Resultsmentioning

confidence: 99%

Genetic Background Modifies Inner Ear and Eye Phenotypes of Jag1 Heterozygous Mice

Kiernan¹,

Hawes

et al. 2007

Genetics

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“…It is not known whether supporting cells in zebrafish also express Notch ligands, although a homologue of Serrate1 may exist (Haddon et al, 1998b). In mammals, the Serrate1 homolog Jagged1 is expressed in supporting cells of the ear, but mouse mutants in Jagged1 indicate that this gene may also have an earlier role in sensory patch specification Tsai et al, 2001, and references therein).…”

Section: Specification Of Hair Cells and Supporting Cells In Sensory mentioning

confidence: 99%

Development of the zebrafish inner ear

Whitfield

Riley

Chiang

et al. 2002

Developmental Dynamics

214

186

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Recent years have seen a renaissance of investigation into the mechanisms of inner ear development. Genetic analysis of zebrafish has contributed significantly to this endeavour, with several dramatic advances reported over the past year or two. Here, we review the major findings from recent work in zebrafish. Several cellular and molecular mechanisms have been elucidated, including the signaling pathways controlling induction of the otic placode, morphogenesis and patterning of the otic vesicle, and elaboration of functional attributes of inner ear.

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“…Two mouse mutants, Headturner and Slalom, with missense mutations in the Notch ligand, Jagged1, have recently been characterized. Homozygotes of both mutants die at early embryonic stages due to vasculature defects, and heterozygotes have an aberrant number of hair cells in the cochlea Tsai et al 2001). Interestingly, Headturner and Slalom are missing one or both of the anterior and posterior ampullae.…”

Section: Notch Signaling Pathwaymentioning

confidence: 99%