Overview

Also Known As:fidget

These mice carry a spontaneous mutation at the Fign locus characterized by head tossing and circular running. Fidget mice also have insufficient growth of the retinal neural epithelium resulting in small eyes, and skeletal abnormalities, including pelvic girdle dysgenesis, skull bone fusions and polydactyly.

Genetic overview

Genetic Background	Generation

a

Allele Type	Gene Symbol	Gene Name
Spontaneous	a	nonagouti

Fign^fi

Allele Type	Gene Symbol	Gene Name
Spontaneous	Fign	fidgetin

stb

Allele Type	Gene Symbol	Gene Name
Spontaneous	stb	stubby

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Research Applications

Developmental Biology Research
Neurobiology Research
Sensorineural Research

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Base Price

Starting at:

$2,854.50 Domestic price Cryo Recovery

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Details

Detailed Description

Mice homozygous for the fidget spontaneous mutation (Fign^fi) toss their heads from side to side and tend to run in circles. Homozygous fidgit mice are hypersensitive to sound in early life, but later lose hearing ability. Corneal lesions are common, and hind foot polydactylism occurs. The bony labyrinth is quite defective. Other abnormalities including small eyes, absence of lachrymal glands, dislocation of the hip, and displaced parafloccular lobes of the cerebellum. Viability of homozygotes is usually less than normal and fertility is poor. The STOCK is also carrying the stubby spontaneous mutation (stb). Homozygous stubby mice are recognizable at 4 or 5 days by a domed head and thick short tail. Adult stubby mice have shorter heads, bodies, and legs compared to wildtype mice. Most females are fertile but males do not breed. Male mice do produce normal numbers of motile sperm. Cartilage histology is within normal limits, but chondrogenesis stops earlier than in normal animals.

Development

The fidget mutation was discovered by Gruneberg in a heterozygous stock as reported in 1943 and eventually obtained by The Jackson Laboratory. The strain was cryopreserved in 1985 and again in 2003.

Control Suggestions

Untyped from the colony

Additional Information

Considerations for Choosing Controls

Genetics

a

Allele Symbol: a

Allele Name	nonagouti
Allele Type	Spontaneous
Allele Synonym(s)
Gene Symbol and Name	a, nonagouti
Gene Synonym(s)
Strain of Origin	old mutant of the mouse fancy
Chromosome	2
General Note	Insertion of the LV30 retrotransposon without the beta4 retrovirus sequence does not cause the nonagouti phenotype. J:278039
Molecular Note	Characterization of this allele shows an insertion of DNA comprised of a 5.5kb virus-like element, VL30, into the first intron of the agouti gene. The VL30 element itself contains an additional 5.5 kb sequence, flanked by 526 bp of direct repeats (beta4 retroviral sequence). The host integration site is the same as for a^t-2Gso and A^w-38J and includes a duplication of four nucleotides of host DNA and a deletion of 2 bp from the end of each repeat. Northern analysis of mRNA from skin of homozygotes shows a smaller agouti message and levels 8 fold lower than found in wild-type.

Fign^fi

Allele Symbol: Fign^fi

Allele Name	fidget
Allele Type	Spontaneous
Allele Synonym(s)	fi
Gene Symbol and Name	Fign, fidgetin
Gene Synonym(s)
Strain of Origin	random-bred
Chromosome	2
Molecular Note	The fidget mutation is due to a retrotransposon insertion in intron 2, interfering with normal RNA processing.

stb

Allele Symbol: stb

Allele Name	stubby
Allele Type	Spontaneous
Allele Synonym(s)
Gene Symbol and Name	stb, stubby
Gene Synonym(s)
Strain of Origin	Not Specified
Chromosome	2

Disease/Phenotype

Disease Terms

Research Areas By Phenotype

This mouse can be used to support research in many areas including:

Genotype: Fign^fi related

Developmental Biology Research
- Neurodevelopmental Defects
- Skeletal Defects
Neurobiology Research
- Neurodevelopmental Defects
- Hearing Defects
- Vestibular Defects
Sensorineural Research
- Eye Defects
- Hearing Defects
- Cataracts
  - nuclear
- Vestibular Defects

Genotype: stb related

Developmental Biology Research
- Growth Defects
- Skeletal Defects
Neurobiology Research
- Neural Tube Defects

Mammalian Phenotype Terms by Genotype

The following phenotype information is associated with a similar, but not exact match to this JAX^® Mice strain

Genotype: Fign^fi/Fign^fi
involves: 129P2/OlaHsd * 129S1/SvImJ * C57BL/6

craniofacial phenotype

cleft secondary palate
- ~19% of Akap8-sufficient, Fign-deficient mice show cleft palate

digestive/alimentary phenotype

cleft secondary palate
- ~19% of Akap8-sufficient, Fign-deficient mice show cleft palate

growth/size/body region phenotype

cleft secondary palate
- ~19% of Akap8-sufficient, Fign-deficient mice show cleft palate

Genotype: stb/stb
Not Specified

limbs/digits/tail phenotype

short tail
- tail length is reduced to 86% of normal

thick tail

short limbs
- limbs are slightly shortened

reproductive system phenotype

abnormal sperm number
- testes that produce no sperm were found in some mutants

reduced male fertility
- most males do not breed

small testis
- testes weight at 29-32 days of age is 57% of normal

skeleton phenotype

abnormal long bone epiphyseal plate morphology

abnormal trabecular bone morphology
- primary trabeculae are shorter, less numerous, and not aligned as well as in controls

decreased long bone epiphyseal plate size
- thinner epiphyseal plate

decreased length of long bones
- limb bone lengths are 85 to 90% of normal

abnormal cartilage development
- chondrogenesis stops earlier than in normal mice

abnormal long bone epiphyseal plate proliferative zone
- proliferating cartilage cell columns are shorter

decreased cranium height
- skull length is 92% of normal

abnormal axial skeleton morphology
- axial skeleton is slightly shortened

decreased width of hypertrophic chondrocyte zone

domed cranium
- domed-shaped head is seen at 4-5 days of age

craniofacial phenotype

domed cranium
- domed-shaped head is seen at 4-5 days of age

decreased cranium height
- skull length is 92% of normal

endocrine/exocrine gland phenotype

small testis
- testes weight at 29-32 days of age is 57% of normal

growth/size/body region phenotype

abnormal body weight
- organ weights at 29-32 days of age 77-92% of normal, except the lungs and kidneys which are comparable in size to controls

decreased body length
- body length is reduced to 89% of normal

abnormal body size
- mutants are shorter and stockier

disproportionate dwarf

postnatal growth retardation
- growth is slightly retarded during the first 4 weeks and adults reach a weight within 3-6 gm of normal siblings

Genotype: Fign^fi/Fign^fi
mixed

cellular phenotype

abnormal cell cycle
- generation time (T) of retinal cells is longer (20 hours) than in wild-type (18 hours); cell cycle duration is increased 2 hours in mutants
- duration of S phase is 8.5 hours compared to 9.75 in wild-type; M phase is shorter (8.5 hours) than in wild-type (9.25 hours

craniofacial phenotype

absent mental foramen
- some mutants show a complete absence of the mental foramina

abnormal craniofacial bone morphology
- decreased size of the pterygoid process and the incidence of the foramen sphenoidale medium
- parieto-squamosal fusion in some
- presphenoid-basisphenoid fusion in some
- interfrontal-frontal fusion in some

absent mandibular canal
- some mutants show a complete absence of the mandibular canal

abnormal mandible morphology

abnormal pterygoid process morphology
- decreased size of the pterygoid process

absent subarcuate fossa
- missing the subarcuate fossa

absent mandibular foramen
- some mutants show a complete absence of the mandibular foramina

endocrine/exocrine gland phenotype

Harderian gland hypertrophy
- hypertrophy of the Harderian glands

absent lacrimal glands
- in both newborn and in adults

Meibomian gland hypertrophy
- hypertrophy of the tarsal glands of the eyelids

growth/size/body region phenotype

decreased body weight
- after first 1-2 weeks, mutants weigh ~50% as much as wild-type littermates

postnatal growth retardation
- during the first 1 or 2 weeks after birth, mice exhibit lag in growth

hearing/vestibular/ear phenotype

absent semicircular canals
- in many animals

deafness
- in subsequent analysis, it was shown that an auditory response could be elicited even in very old mutant mice
- from 3-4 months of age, mice have severe hearing loss or are completely deaf

abnormal otic capsule morphology
- missing the fenestra flocculi in the auditory capsule

increased susceptibility to age-related hearing loss
- mutants initially display hypersensitivity to sound at 3 weeks of age, but this is followed by a period where they respond to only drastic stimuli
- from 3-4 months of age onward, no response to auditory stimuli is observed

absent lateral semicircular canal
- always completely absent

abnormal membranous labyrinth morphology
- the dorsal part of membranous labyrinth fails to differentiate into normal semicircular canals during embryonic development

immune system phenotype

conjunctivitis
- after birth once the eyes have opened, discharge from the conjunctiva occurs, such that the eyelids stick together; one or both eyes are closed in affected animals for much of their lives

eye inflammation
- blisters of the corneal epithelium are present at early ages; these burst and coalesce, sometimes forming large ulcers

integument phenotype

Meibomian gland hypertrophy
- hypertrophy of the tarsal glands of the eyelids

limbs/digits/tail phenotype

polydactyly
- doubling of digits tends to disappear with growth
- usually involving first digit of hindlimbs, observed in small subset of animals
- occasionally only doubling of distal pad of toe, with or without claw occurs; in some cases, extra toe is completely separated from original and larger in size

hip dislocation
- Background Sensitivity - in some mutant mice of GFF background had a complete dislocation of the hip

fused tarsal bones
- significantly higher incidence of tarsal fusions

mammalian phenotype

hearing/vestibular/ear phenotype
- Normal - do not turn deaf in old age
- Normal - the cochlea appears normal

nervous system phenotype

abnormal cerebellar lobule formation
- the parafloccular lobe of the cerebellum becomes very abnormal in shape

reproductive system phenotype

reduced fertility
- females are sometimes sterile while others show reduced fertility

skeleton phenotype

abnormal craniofacial bone morphology
- decreased size of the pterygoid process and the incidence of the foramen sphenoidale medium
- parieto-squamosal fusion in some
- presphenoid-basisphenoid fusion in some
- interfrontal-frontal fusion in some

absent mandibular canal
- some mutants show a complete absence of the mandibular canal

absent mental foramen
- some mutants show a complete absence of the mental foramina

abnormal pterygoid process morphology
- decreased size of the pterygoid process

absent subarcuate fossa
- missing the subarcuate fossa

abnormal pelvic girdle bone morphology
- considerably reduced anterior iliac spine

abnormal mandible morphology

absent mandibular foramen
- some mutants show a complete absence of the mandibular foramina

fused tarsal bones
- significantly higher incidence of tarsal fusions

abnormal acetabular fossa morphology
- a decrease in the depth and in the diameter of the fossa acetabuli

hip dislocation
- Background Sensitivity - in some mutant mice of GFF background had a complete dislocation of the hip

vision/eye phenotype

small lens
- reduced in size from early stages of embryogenesis
- approximately 80% of the normal size in length

absent lacrimal glands
- in both newborn and in adults

eye inflammation
- blisters of the corneal epithelium are present at early ages; these burst and coalesce, sometimes forming large ulcers

abnormal retinal development
- retina matures more slowly than in wild-type

conjunctivitis
- after birth once the eyes have opened, discharge from the conjunctiva occurs, such that the eyelids stick together; one or both eyes are closed in affected animals for much of their lives

Meibomian gland hypertrophy
- hypertrophy of the tarsal glands of the eyelids

abnormal cornea morphology
- small perforation of the entire thickness of each cornea near its centre in a few mutants

abnormal retinal progenitor cell morphology
- cells in the retinal anlage do not begin transition to differentiated state in general nuclear layer until E12, whereas wild-type cells start transition at E11

corneal opacity
- occurs in later stages, sometimes covering cornea completely

corneal vascularization
- occurs in later stages of inflammation

microphthalmia
- much reduced eyes from the age of 13 days onward

behavior/neurological phenotype

increased startle reflex
- mutants initially display hypersensitivity to sound at 3 weeks of age

abnormal locomotor coordination
- when suspended by the tail, mice exhibit violent jerking movements of their bodies without purposeful coordination
- when placed in water, mice appear to lose all sense of direction and begin to gyrate frantically

abnormal maternal nurturing
- some females produce litters which they fail to rear

head shaking
- affected mice shake their heads from side to side, from a few times to many times in rapid succession, starting as early as 3-4 days after birth; excursions are small
- shaking is most marked when mouse is active and ambulatory; movements tend to decrease when animal is quiet

impaired coordination
- when suspended by the tail, mice exhibit violent jerking movements of their bodies without purposeful coordination

bidirectional circling
- animals circle, but behavior is not marked as in other circling mutants such as waltzers

cardiovascular system phenotype

corneal vascularization
- occurs in later stages of inflammation

Genotype: Fign^fi/Fign^fi
involves: 129S1/SvImJ * C57BL/6

craniofacial phenotype

cleft secondary palate
- some mice show complete cleft of secondary palate

digestive/alimentary phenotype

cleft secondary palate
- some mice show complete cleft of secondary palate

growth/size/body region phenotype

cleft secondary palate
- some mice show complete cleft of secondary palate

References

Additional References

Lane PW; Dickie MM. 1968. Three recessive mutations producing disproportionate dwarfing in mice: achondroplasia, brachymorphic, and stubby. J Hered 59(5):300-8PubMed: 5713631 MGI: J:5109
Miller WA; Flynn-Miller KL. 1976. A chondroplastic, brachymorphic and stubby chondrodystophies in mice. J Comp Pathol 86(3):349-63PubMed: 939824 MGI: J:5669

Additional - a related

Additional - Fign^fi related

Additional - stb related

Technical Support

CONTACT TECHNICAL SUPPORT

Genotyping Protocols
- Genotyping resources and troubleshooting
Breeding Considerations

When maintaining a live colony, mice can be bred, with progeny testing, heterozygote x heterozygote for stb in repulsion with fi, and heterozygote x heterozygote for fi in repulsion with stb . These genes are linked on chromosome 2 in the following order: stb Fign^fi a. Homozygous stb males are sterile. There is decreased viability in mice homozygous for Fign^fi. Homozygous Fign^fi males may need to be removed from the cage when the female is pregnant.
- Additional Breeding and Husbandry Support
Appearance
- black
  Related Genotype: a/a
Citation
When using the fidget mouse strain in a publication, please cite the originating article(s) and include JAX stock #000312 in your Materials and Methods section.

Animal Health Reports

Facility Barrier Level Descriptions

Production of mice from cryopreserved embryos or sperm occurs in a maximum barrier room, G200

Pricing & Availability

Cryo Recovery

Domestic
International

Live Mouse

Age

Genotype

Price

weeks

Cryorecovery - Pricing

Service/Product	Description	Price
> >	Heterozygous for stb, Heterozygous for Fign<fi>, Homozygous for a

Payment Terms and Conditions

Terms are granted by individual review and stated on the customer invoice(s) and account statement. These transactions are payable in U.S. currency within the granted terms. Payment for services, products, shipping containers, and shipping costs that are rendered are expected within the payment terms indicated on the invoice or stated by contract. Invoices and account balances in arrears of stated terms may result in The Jackson Laboratory pursuing collection activities including but not limited to outside agencies and court filings.

The Jackson Laboratory's Genotype Promise

The Jackson Laboratory has rigorous genetic quality control and mutant gene genotyping programs to ensure the genetic background of JAX® Mice strains as well as the genotypes of strains with identified molecular mutations. JAX® Mice strains are only made available to researchers after meeting our standards. However, the phenotype of each strain may not be fully characterized and/or captured in the strain data sheets. Therefore, we cannot guarantee a strain's phenotype will meet all expectations. To ensure that JAX® Mice will meet the needs of individual research projects or when requesting a strain that is new to your research, we suggest ordering and performing tests on a small number of mice to determine suitability for your particular project. We do not guarantee breeding performance and therefore suggest that investigators order more than one breeding pair to avoid delays in their research.

Terms Of Use

General Terms and Conditions

Questions about Terms of Use

Licensing Information

Phone: 207-288-6470

Email: TechTran@jax.org

Also Known As:fidget

Genetic overview

Research Applications

Base Price

Detailed Description

Development

Control Suggestions

Additional Information

a

Allele Symbol: a

Fignfi

Allele Symbol: Fignfi

stb

Allele Symbol: stb

Disease Terms

Research Areas By Phenotype

This mouse can be used to support research in many areas including:

Genotype: Fignfi related

Genotype: stb related

Mammalian Phenotype Terms by Genotype

Genotype: Fignfi/Fignfiinvolves: 129P2/OlaHsd * 129S1/SvImJ * C57BL/6

craniofacial phenotype

digestive/alimentary phenotype

growth/size/body region phenotype

Genotype: stb/stbNot Specified

limbs/digits/tail phenotype

reproductive system phenotype

skeleton phenotype

craniofacial phenotype

endocrine/exocrine gland phenotype

growth/size/body region phenotype

Genotype: Fignfi/Fignfimixed

cellular phenotype

craniofacial phenotype

endocrine/exocrine gland phenotype

growth/size/body region phenotype

hearing/vestibular/ear phenotype

immune system phenotype

integument phenotype

limbs/digits/tail phenotype

mammalian phenotype

nervous system phenotype

reproductive system phenotype

skeleton phenotype

vision/eye phenotype

behavior/neurological phenotype

cardiovascular system phenotype

Genotype: Fignfi/Fignfiinvolves: 129S1/SvImJ * C57BL/6

craniofacial phenotype

digestive/alimentary phenotype

growth/size/body region phenotype

References

Additional References

Additional - a related

Additional - Fignfi related

Additional - stb related

Genotyping Protocols

Breeding Considerations

Appearance

Citation

Animal Health Reports

Production of mice from cryopreserved embryos or sperm occurs in a maximum barrier room, G200

Live Mouse

Cryorecovery - Pricing

Payment Terms and Conditions

The Jackson Laboratory's Genotype Promise

Terms Of Use

Licensing Information

Fign^fi

Allele Symbol: Fign^fi

Genotype: Fign^fi related

Genotype: Fign^fi/Fign^fi
involves: 129P2/OlaHsd * 129S1/SvImJ * C57BL/6

Genotype: stb/stb
Not Specified

Genotype: Fign^fi/Fign^fi
mixed

Genotype: Fign^fi/Fign^fi
involves: 129S1/SvImJ * C57BL/6

Additional - Fign^fi related