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Phenotypes associated with this allele
Allele Symbol
Allele Name
Allele ID
FuzGt(OSTGST001398)Lex
gene trap OSTGST001398, Lexicon Genetics
MGI:3531090
Summary 2 genotypes
Jump to Allelic Composition Genetic Background Genotype ID
hm1
FuzGt(OSTGST001398)Lex/FuzGt(OSTGST001398)Lex involves: 129S5/SvEvBrd * C57BL/6 MGI:5297069
hm2
FuzGt(OSTGST001398)Lex/FuzGt(OSTGST001398)Lex involves: 129S5/SvEvBrd * C57BL/6N MGI:4438900


Genotype
MGI:5297069
hm1
Allelic
Composition
FuzGt(OSTGST001398)Lex/FuzGt(OSTGST001398)Lex
Genetic
Background
involves: 129S5/SvEvBrd * C57BL/6
Find Mice Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
FuzGt(OSTGST001398)Lex mutation (0 available); any Fuz mutation (23 available)
phenotype observed in females
phenotype observed in males
N normal phenotype

Craniofacial defects in FuzGt(OSTGST001398)Lex/FuzGt(OSTGST001398)Lex mice

mortality/aging
• die immediately after birth

craniofacial
• defects in the nasal cavity, facial bones and craniofacial cartilage
• at E14.5, elongates dorsoventrally and expands the dorsal end along the median-lateral axis
• at E16.0 the anterior cartilage is malformed with an ascending branch and reduced ossification around the cartilage
• increased proliferation of cells at E14.5
• hyperplastic and malformed at E18.5
• at E18.5
• fail to form incisor tooth buds
• upper and lower incisor initiation does not occur
• however, molar buds do form
• at E14.5 the mandible is thickened in the dorsal-ventral axis and shortened in the anterior-posterior axis
• at E18.5, the tongue muscles appear to be fused with the mandible and there is an abnormal ventral bulge in the mandible
• at E18.5
• at E18.5
• abnormal growth of palate tissue at E14.5
• descending palate shelves at E16.5 suggesting a delay in elevation
• at E14.5 the tongue has a rudimentary root structure but fails to elongate in the anterior-posterior axis
• at E18.5, the tongue muscles appear to be fused with the mandible

nervous system
• many brain structures are undeveloped at E14.5
• the corpus striatum mediale is displaced at E12.5
• absence of the optic recess at E12.5
• the trigeminal nerve appears to replace Rathke's pouch

vision/eye
• at E18.5

hearing/vestibular/ear
• displaced at E12.5

endocrine/exocrine glands
• the trigeminal nerve appears to replace Rathke's pouch

digestive/alimentary system
• abnormal growth of palate tissue at E14.5
• descending palate shelves at E16.5 suggesting a delay in elevation
• at E14.5 the tongue has a rudimentary root structure but fails to elongate in the anterior-posterior axis
• at E18.5, the tongue muscles appear to be fused with the mandible

skeleton
• at E14.5, elongates dorsoventrally and expands the dorsal end along the median-lateral axis
• at E16.0 the anterior cartilage is malformed with an ascending branch and reduced ossification around the cartilage
• increased proliferation of cells at E14.5
• hyperplastic and malformed at E18.5
• at E18.5
• fail to form incisor tooth buds
• upper and lower incisor initiation does not occur
• however, molar buds do form
• at E14.5 the mandible is thickened in the dorsal-ventral axis and shortened in the anterior-posterior axis
• at E18.5, the tongue muscles appear to be fused with the mandible and there is an abnormal ventral bulge in the mandible
• at E18.5
• at E18.5
• delay in ossification of the sphenoid and basioccipital bones at E16.0
• membranous ossification of the premaxilla, maxilla, mandible, frontal and parietal bones is missing at E16.0

embryo
• at E14.5 the amount of primary cilium is significantly decreased in the mandibular mesenchyme

growth/size/body
• at E18.5
• fail to form incisor tooth buds
• upper and lower incisor initiation does not occur
• however, molar buds do form
• abnormal growth of palate tissue at E14.5
• descending palate shelves at E16.5 suggesting a delay in elevation
• at E14.5 the tongue has a rudimentary root structure but fails to elongate in the anterior-posterior axis
• at E18.5, the tongue muscles appear to be fused with the mandible
• at E14.5 the amount of primary cilium is significantly decreased in the mandibular mesenchyme




Genotype
MGI:4438900
hm2
Allelic
Composition
FuzGt(OSTGST001398)Lex/FuzGt(OSTGST001398)Lex
Genetic
Background
involves: 129S5/SvEvBrd * C57BL/6N
Find Mice Using the International Mouse Strain Resource (IMSR)
Mouse lines carrying:
FuzGt(OSTGST001398)Lex mutation (0 available); any Fuz mutation (23 available)
phenotype observed in females
phenotype observed in males
N normal phenotype
cardiovascular system
• mice exhibit conotruncal defects unlike wild-type mice
• some mice exhibit ventral septal defects unlike wild-type mice
• some mice exhibit single outflow tracks unlike wild-type mice

skeleton
• primary ciliogenesis in Meckel's cartilage is abnormal compared to in wild-type mice with shorter cilia

nervous system
• even mice with mild overt neural tube closure defects had severely dilated brain ventricles
• in some mice at E18.5
• in some mice at E18.5

limbs/digits/tail
• on all limbs

vision/eye

craniofacial
• primary ciliogenesis in Meckel's cartilage is abnormal compared to in wild-type mice with shorter cilia

respiratory system





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last database update
04/23/2024
MGI 6.23
The Jackson Laboratory