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Type Mutant Stock; Targeted Mutation; Additional information on Genetically Engineered and Mutant Mice. Visit our online Nomenclature tutorial. Species laboratory mouse Generation F?+2
Generation DefinitionsDonating Investigator Dr. Martin Matzuk, Baylor College of Medicine Description
Homozygous mice die within hours after birth due to a failure to breathe. They are smaller in size than normal wildtype siblings and show craniofacial, musculoskeletal, and skin defects.
| Control | ||
|---|---|---|
| Wild-type from the colony | ||
| 101045 B6129SF2/J | (approximate) | |
| Considerations for Choosing Controls | ||
Facebase: models
View Facebase: models (58 strains)
View Related Disease (OMIM) Terms
Related Disease (OMIM) Terms provided by MGI
- Model with phenotypic similarity to human disease where etiologies are distinct. Human genes are associated with this disease. Orthologs of these genes do not appear in the mouse genotype(s).
Restrictive Dermopathy, Lethal
View Mammalian Phenotype Terms
Mammalian Phenotype Terms provided by MGI
assigned by genotype
Fsttm1Zuk/Fst+
involves: 129S7/SvEvBrd * C57BL/6
- skeleton phenotype
- abnormal rib development
- in 17% (6 of 35) heterozygotes, development of the thirteenth pair of ribs is limited; not oberved in wild-type controls (MGI Ref ID J:23925)
- a higher percentage of heterozygotes show limited formation of the thirteenth rib pair on an inbred 129/SvEv background than on a hybrid 129/SvEv x C57BL/6 background (26.6% vs 17%, respectively) (MGI Ref ID J:23925)
- decreased lumbar vertebrae number
- 60% (21 of 35) heterozygotes have five lumbar vertebrae instead of six; only 2 of 15 wild-type controls show this defect (MGI Ref ID J:23925)
- a higher percentage of heterozygotes show five lumbar vertebrae on an inbred 129/SvEv background than on a hybrid 129/SvEv x C57BL/6 background (86.6% vs 60%, respectively) (MGI Ref ID J:23925)
Fsttm1Zuk/Fsttm1Zuk
involves: 129S7/SvEvBrd * C57BL/6
- mortality/aging
- complete neonatal lethality
- homozygotes survive to birth but die within a few hours of birth due to respiratory failure (MGI Ref ID J:23925)
- craniofacial phenotype
- absent hard palate
- 21.4% (6 of 28) newborn homozygotes lack a hard palate (MGI Ref ID J:23925)
- absent lower incisors
- 6 of 34 newborn homozygotes lack lower incisors (MGI Ref ID J:23925)
- cleft secondary palate
- 15.8% (3 of 19) newborn homozygotes display a cleft secondary palate (MGI Ref ID J:23925)
- growth retardation of incisors
- 92% (23 of 34) newborn homozygotes show delayed development of lower incisors (MGI Ref ID J:23925)
- growth/size phenotype
- decreased fetal size
- at E18.5, homozygotes are smaller than wild-type counterparts (MGI Ref ID J:23925)
- decreased fetal weight
- at E18.5, homozygotes weigh ~12% less than wild-type counterparts (MGI Ref ID J:23925)
- fetal growth retardation
- newborn homozygotes are growth retarded (MGI Ref ID J:23925)
- muscle phenotype
- decreased skeletal muscle mass
- skeleton phenotype
- abnormal rib development
- in 61% (17 of 28) homozygotes, development of the thirteenth pair of ribs is limited (MGI Ref ID J:23925)
- a higher percentage of homozygotes show limited formation of the thirteenth rib pair on an inbred 129/SvEv background than on a hybrid 129/SvEv x C57BL/6 background (81.8% vs 61%, respectively) (MGI Ref ID J:23925)
- abnormal rib-sternum attachment
- in 4 of 28 homozygotes, one or both of the seventh ribs fail to fuse to the sternum (MGI Ref ID J:23925)
- decreased lumbar vertebrae number
- all (28 of 28) newborn homozygotes have five lumbar vertebrae instead of six; only 2 of 15 wild-type controls show this defect (MGI Ref ID J:23925)
- decreased rib number
- in 28.6% (8 of 28) homozygotes, the thirteenth pair of ribs is absent (MGI Ref ID J:23925)
- digestive/alimentary phenotype
- absent hard palate
- 21.4% (6 of 28) newborn homozygotes lack a hard palate (MGI Ref ID J:23925)
- cleft secondary palate
- 15.8% (3 of 19) newborn homozygotes display a cleft secondary palate (MGI Ref ID J:23925)
- homeostasis/metabolism phenotype
- cyanosis
- newborn homozygotes appear cyanotic (MGI Ref ID J:23925)
- respiratory system phenotype
- respiratory failure
- integument phenotype
- abnormal vibrissa morphology
- mutant whiskers are very thin and incorrectly oriented with shafts projecting parallel before turning perpendicularly (MGI Ref ID J:23925)
- hyperkeratosis
- newborns display a 25% increase in the stratum corneum cells relative to wild-type controls (MGI Ref ID J:23925)
- pallor
- newborn homozygotes appear hypoxic (pale) (MGI Ref ID J:23925)
- shiny skin
- newborn homozygotes display a shiny skin (MGI Ref ID J:23925)
- thick epidermis
- newborn homozygotes show a thickened granular and stratum corneum (MGI Ref ID J:23925)
- tight skin
- newborn homozygotes display a taut skin (MGI Ref ID J:23925)
Fsttm1Zuk/Fsttm1Zuk
B6.129S7-Fsttm1Zuk
- nervous system phenotype
- abnormal neuron differentiation
- homozygotes display decreased olfactory epithelium neurogenesis, as shown by significantly reduced neurogenin-1-expressing immediate neuronal precursors and Ncam1-expressing olfactory receptor neurons (MGI Ref ID J:81451)
- decreased neuronal precursor cell number (MGI Ref ID J:81451)
- abnormal olfactory neuron morphology
- at E17.5, homozygotes display a 37% decrease in proliferating, neurogenin-1-expressing immediate neuronal precursors within the olfactory epithelium (MGI Ref ID J:81451)
- taste/olfaction phenotype
- abnormal olfactory epithelium morphology
- homozygotes display a 38% decrease in thickness of the olfactory epithelium (MGI Ref ID J:81451)
- vision/eye phenotype
- thin retinal ganglion layer
- mutant retinas exhibit a 26% reduction in the number of cells in the ganglion cell layer and a marked decrease in thickness of the differentiated ganglion cell layer (MGI Ref ID J:99244)
- unlike the situation in olfactory epithelium, mutant retinas display normal overall thickness as well as normal progenitor cell proliferation (MGI Ref ID J:99244)
- in addition, mutant retinas show no obvious differences in amacrine cell or photoreceptor production relative to wild-type (MGI Ref ID J:99244)
- respiratory system phenotype
- abnormal olfactory epithelium morphology
- homozygotes display a 38% decrease in thickness of the olfactory epithelium (MGI Ref ID J:81451)
- craniofacial phenotype
- abnormal olfactory epithelium morphology
- homozygotes display a 38% decrease in thickness of the olfactory epithelium (MGI Ref ID J:81451)
- cellular phenotype
- abnormal neuron differentiation
- homozygotes display decreased olfactory epithelium neurogenesis, as shown by significantly reduced neurogenin-1-expressing immediate neuronal precursors and Ncam1-expressing olfactory receptor neurons (MGI Ref ID J:81451)
- decreased neuronal precursor cell number (MGI Ref ID J:81451)
The following phenotype information may relate to a genetic background differing from this JAX® Mice strain.
Fsttm1Zuk/Fst+
involves: 129S7/SvEvBrd
- skeleton phenotype
- abnormal rib development
- in 26.6% (4 of 15) heterozygotes, development of the thirteenth pair of ribs is limited; not oberved in wild-type controls (MGI Ref ID J:23925)
- a higher percentage of heterozygotes show limited formation of the thirteenth rib pair on an inbred 129/SvEv background than on a hybrid 129/SvEv x C57BL/6 background (26.6% vs 17%, respectively) (MGI Ref ID J:23925)
- decreased lumbar vertebrae number
- 86.6% (13 of 15) heterozygotes have five lumbar vertebrae instead of six found in wild-type controls (MGI Ref ID J:23925)
- a higher percentage of heterozygotes show five lumbar vertebrae on an inbred 129/SvEv background than on a hybrid 129/SvEv x C57BL/6 background (86.6% vs 60%, respectively) (MGI Ref ID J:23925)
Fsttm1Zuk/Fst+
B6.129S7-Fsttm1Zuk
- muscle phenotype
- abnormal extensor digitorum longus morphology
- increase in the percentage of oxidative type I fibers (MGI Ref ID J:182827)
- abnormal gastrocnemius morphology
- increase in the number of oxidative type I fibers (MGI Ref ID J:182827)
- decreased gastrocnemius weight (MGI Ref ID J:182827)
- abnormal muscle physiology
- abnormal pectoral muscle morphology
- decreased weight (MGI Ref ID J:182827)
- abnormal skeletal muscle fiber morphology
- increase in the proportion of smaller fibers that stain more darkly in hematoxylin and eosin stained sections corresponding to mixed glycolytic/oxidative type IIa fibers (MGI Ref ID J:182827)
- shift towards more oxidative fibers (MGI Ref ID J:182827)
- decrease in mean fiber diameter for each fiber type (MGI Ref ID J:182827)
- decreased quadriceps weight (MGI Ref ID J:182827)
- decreased skeletal muscle weight
- homeostasis/metabolism phenotype
- increased physiological sensitivity to xenobiotic
- impaired muscle regeneration and enhanced fibrosis in the gastrocnemius muscle following cardiotoxin-induced injury (MGI Ref ID J:182827)
Fsttm1Zuk/Fsttm1Zuk
involves: 129S7/SvEvBrd
- mortality/aging
- complete neonatal lethality
- homozygotes survive to birth but die within a few hours of birth due to respiratory failure (MGI Ref ID J:23925)
- craniofacial phenotype
- absent hard palate
- 55% (6 of 11) newborn homozygotes lack a hard palate (MGI Ref ID J:23925)
- growth retardation of incisors
- all newborn homozygotes (11 of 11) show delayed development of lower incisors (MGI Ref ID J:23925)
- growth/size phenotype
- decreased fetal size
- at E18.5, homozygotes are smaller than wild-type counterparts (MGI Ref ID J:23925)
- decreased fetal weight
- at E18.5, homozygotes weigh ~12% less than wild-type counterparts (MGI Ref ID J:23925)
- fetal growth retardation
- newborn homozygotes are growth retarded (MGI Ref ID J:23925)
- muscle phenotype
- decreased skeletal muscle mass
- skeleton phenotype
- abnormal rib development
- in 81.8% (9 of 11) homozygotes, development of the thirteenth pair of ribs is limited (MGI Ref ID J:23925)
- a higher percentage of homozygotes show limited formation of the thirteenth rib pair on an inbred 129/SvEv background than on a hybrid 129/SvEv x C57BL/6 background (81.8% vs 61%, respectively) (MGI Ref ID J:23925)
- decreased lumbar vertebrae number
- all (11 of 11) newborn homozygotes have five lumbar vertebrae instead of six found in wild-type mice (MGI Ref ID J:23925)
- decreased rib number
- in 2 of 11 homozygotes, the thirteenth pair of ribs is absent (MGI Ref ID J:23925)
- digestive/alimentary phenotype
- absent hard palate
- 55% (6 of 11) newborn homozygotes lack a hard palate (MGI Ref ID J:23925)
- homeostasis/metabolism phenotype
- cyanosis
- newborn homozygotes appear cyanotic (MGI Ref ID J:23925)
- respiratory system phenotype
- respiratory failure
- integument phenotype
- abnormal vibrissa morphology
- mutant whiskers are very thin and incorrectly oriented with shafts projecting parallel before turning perpendicularly (MGI Ref ID J:23925)
- hyperkeratosis
- newborns display a 25% increase in the stratum corneum cells relative to wild-type controls (MGI Ref ID J:23925)
- pallor
- newborn homozygotes appear hypoxic (pale) (MGI Ref ID J:23925)
- shiny skin
- newborn homozygotes display a shiny skin (MGI Ref ID J:23925)
- thick epidermis
- newborn homozygotes show a thickened granular and stratum corneum (MGI Ref ID J:23925)
- tight skin
- newborn homozygotes display a taut skin (MGI Ref ID J:23925)
View Research Applications
Research Applications
This mouse can be used to support research in many areas including:Fsttm1Zuk related
Dermatology Research
Skin and Hair Texture Defects
Developmental Biology Research
Craniofacial and Palate Defects
Embryonic Lethality (Homozygous)
Growth Defects
Skeletal Defects
Reproductive Biology Research
Endocrine Deficiencies Affecting Gonads
Fertility Defects
| Allele Symbol | Fsttm1Zuk | ||
|---|---|---|---|
| Allele Name | targeted mutation 1, Martin M Matzuk | ||
| Allele Type | Targeted (knock-out) | ||
| Common Name(s) | Fst-; Fsttm1; fsm1; | ||
| Mutation Made By | Dr. Martin Matzuk, Baylor College of Medicine | ||
| Strain of Origin | 129S7/SvEvBrd-Hprt | ||
| ES Cell Line Name | AB2.1 | ||
| ES Cell Line Strain | 129S7/SvEvBrd-Hprt | ||
| Gene Symbol and Name | Fst, follistatin | ||
| Chromosome | 13 | ||
| Gene Common Name(s) | FOL1; FS; Fst-288; RATFOL1; | ||
| Molecular Note | An Hprt expression cassette replaced the entire gene, including all 6 exons. [MGI Ref ID J:23925] | ||
Genotyping Protocols
Generic HPRT_Alternative,Separated MCA
Generic HPRT_Alternative, Separated PCR
Human HPRT/KO2, Separated PCR
Helpful Links
Genotyping resources and troubleshooting
Matzuk MM; Lu N; Vogel H; Sellheyer K; Roop DR; Bradley A. 1995. Multiple defects and perinatal death in mice deficient in follistatin [see comments] Nature 374(6520):360-3. [PubMed: 7885475] [MGI Ref ID J:23925]
Fsttm1Zuk relatedAntsiferova M; Klatte JE; Bodo E; Paus R; Jorcano JL; Matzuk MM; Werner S; Kogel H. 2009. Keratinocyte-derived follistatin regulates epidermal homeostasis and wound repair. Lab Invest 89(2):131-41. [PubMed: 19079322] [MGI Ref ID J:144223]
Beites CL; Hollenbeck PL; Kim J; Lovell-Badge R; Lander AD; Calof AL. 2009. Follistatin modulates a BMP autoregulatory loop to control the size and patterning of sensory domains in the developing tongue. Development 136(13):2187-97. [PubMed: 19474151] [MGI Ref ID J:149988]
Gokoffski KK; Wu HH; Beites CL; Kim J; Kim EJ; Matzuk MM; Johnson JE; Lander AD; Calof AL. 2011. Activin and GDF11 collaborate in feedback control of neuroepithelial stem cell proliferation and fate. Development 138(19):4131-42. [PubMed: 21852401] [MGI Ref ID J:176045]
Jhaveri S; Erzurumlu RS; Chiaia N; Kumar TR; Matzuk MM. 1998. Defective whisker follicles and altered brainstem patterns in activin and follistatin knockout mice. Mol Cell Neurosci 12(4-5):206-19. [PubMed: 9828086] [MGI Ref ID J:50975]
Jorgez CJ; Klysik M; Jamin SP; Behringer RR; Matzuk MM. 2004. Granulosa cell-specific inactivation of follistatin causes female fertility defects. Mol Endocrinol 18(4):953-67. [PubMed: 14701941] [MGI Ref ID J:89081]
Jukkola T; Lahti L; Naserke T; Wurst W; Partanen J. 2006. FGF regulated gene-expression and neuronal differentiation in the developing midbrain-hindbrain region. Dev Biol 297(1):141-57. [PubMed: 16782087] [MGI Ref ID J:112633]
Kim J; Wu HH; Lander AD; Lyons KM; Matzuk MM; Calof AL. 2005. GDF11 controls the timing of progenitor cell competence in developing retina. Science 308(5730):1927-30. [PubMed: 15976303] [MGI Ref ID J:99244]
Klein OD; Lyons DB; Balooch G; Marshall GW; Basson MA; Peterka M; Boran T; Peterkova R; Martin GR. 2008. An FGF signaling loop sustains the generation of differentiated progeny from stem cells in mouse incisors. Development 135(2):377-85. [PubMed: 18077585] [MGI Ref ID J:130572]
Lee SJ; Lee YS; Zimmers TA; Soleimani A; Matzuk MM; Tsuchida K; Cohn RD; Barton ER. 2010. Regulation of muscle mass by follistatin and activins. Mol Endocrinol 24(10):1998-2008. [PubMed: 20810712] [MGI Ref ID J:182827]
Lin SY; Craythorn RG; O'Connor AE; Matzuk MM; Girling JE; Morrison JR; de Kretser DM. 2008. Female infertility and disrupted angiogenesis are actions of specific follistatin isoforms. Mol Endocrinol 22(2):415-29. [PubMed: 17932109] [MGI Ref ID J:130061]
Nakamura M; Matzuk MM; Gerstmayer B; Bosio A; Lauster R; Miyachi Y; Werner S; Paus R. 2003. Control of pelage hair follicle development and cycling by complex interactions between follistatin and activin. FASEB J 17(3):497-9. [PubMed: 12514121] [MGI Ref ID J:118511]
Pangas SA. 2012. Regulation of the ovarian reserve by members of the transforming growth factor beta family. Mol Reprod Dev 79(10):666-79. [PubMed: 22847922] [MGI Ref ID J:190579]
Roy A; Matzuk MM. 2006. Deconstructing mammalian reproduction: using knockouts to define fertility pathways. Reproduction 131(2):207-19. [PubMed: 16452715] [MGI Ref ID J:108425]
Wang XP; Suomalainen M; Jorgez CJ; Matzuk MM; Wankell M; Werner S; Thesleff I. 2004. Modulation of activin/bone morphogenetic protein signaling by follistatin is required for the morphogenesis of mouse molar teeth. Dev Dyn 231(1):98-108. [PubMed: 15305290] [MGI Ref ID J:91704]
Wang XP; Suomalainen M; Jorgez CJ; Matzuk MM; Werner S; Thesleff I. 2004. Follistatin regulates enamel patterning in mouse incisors by asymmetrically inhibiting BMP signaling and ameloblast differentiation. Dev Cell 7(5):719-30. [PubMed: 15525533] [MGI Ref ID J:94518]
Wu HH; Ivkovic S; Murray RC; Jaramillo S; Lyons KM; Johnson JE; Calof AL. 2003. Autoregulation of Neurogenesis by GDF11. Neuron 37(2):197-207. [PubMed: 12546816] [MGI Ref ID J:81451]
Yao HH; Aardema J; Holthusen K. 2006. Sexually dimorphic regulation of inhibin Beta B in establishing gonadal vasculature in mice. Biol Reprod 74(5):978-83. [PubMed: 16452457] [MGI Ref ID J:107812]
Yao HH; Matzuk MM; Jorgez CJ; Menke DB; Page DC; Swain A; Capel B. 2004. Follistatin operates downstream of Wnt4 in mammalian ovary organogenesis. Dev Dyn 230(2):210-5. [PubMed: 15162500] [MGI Ref ID J:90277]
Animal Health Reports
Production of mice from cryopreserved embryos or sperm occurs in a maximum barrier room, G200.
| Pricing for USA, Canada and Mexico shipping destinations |
|
Cryopreserved Mice - Ready for Recovery
Animals Provided
Price (US dollars $) Cryorecovery* $3000.00 At least two mice that carry the mutation (if it is a mutant strain) will be provided. Their genotypes may not reflect those discussed in the strain description. Please inquire for possible genotypes and see additional details below.
Standard Supply
Cryopreserved. Ready for recovery. Please refer to pricing and supply notes on the strain data sheet for further information.
Supply Notes
- Cryorecovery - Standard.
Progeny testing is not required.
The average number of mice provided from recovery of our cryopreserved strains is 10. The total number of animals provided, their gender and genotype will vary. We will fulfill your order by providing at least two pair of mice, at least one animal of each pair carrying the mutation of interest. Please inquire if larger numbers of animals with specific genotype and genders are needed. Animals typically ship between 11 and 14 weeks from the date of your order. If a second cryorecovery is needed in order to provide the minimum number of animals, animals will ship within 25 weeks. IMPORTANT NOTE: The genotypes of animals provided may not reflect the mating scheme utilized by The Jackson Laboratory prior to cryopreservation, or that discussed in the strain description. Please inquire about possible genotypes which will be recovered for this specific strain. The Jackson Laboratory cannot guarantee the reproductive success of mice shipped to your facility. If the mice are lost after the first three days (post-arrival) or do not produce progeny at your facility, a new order and fee will be necessary.Cryorecovery to establish a Dedicated Supply for greater quantities of mice.
Mice recovered can be used to establish a dedicated colony to contractually supply you mice according to your requirements. Price by quotation. For more information on Dedicated Supply, please contact JAX® Services, Tel: 1-800-422-6423 (from U.S.A., Canada or Puerto Rico only) or 1-207-288-5845 (from any location).
| Pricing for International shipping destinations |
|
Cryopreserved Mice - Ready for Recovery
Animals Provided
Price (US dollars $) Cryorecovery* $3900.00 At least two mice that carry the mutation (if it is a mutant strain) will be provided. Their genotypes may not reflect those discussed in the strain description. Please inquire for possible genotypes and see additional details below.
Standard Supply
Cryopreserved. Ready for recovery. Please refer to pricing and supply notes on the strain data sheet for further information.
Supply Notes
- Cryorecovery - Standard.
Progeny testing is not required.
The average number of mice provided from recovery of our cryopreserved strains is 10. The total number of animals provided, their gender and genotype will vary. We will fulfill your order by providing at least two pair of mice, at least one animal of each pair carrying the mutation of interest. Please inquire if larger numbers of animals with specific genotype and genders are needed. Animals typically ship between 11 and 14 weeks from the date of your order. If a second cryorecovery is needed in order to provide the minimum number of animals, animals will ship within 25 weeks. IMPORTANT NOTE: The genotypes of animals provided may not reflect the mating scheme utilized by The Jackson Laboratory prior to cryopreservation, or that discussed in the strain description. Please inquire about possible genotypes which will be recovered for this specific strain. The Jackson Laboratory cannot guarantee the reproductive success of mice shipped to your facility. If the mice are lost after the first three days (post-arrival) or do not produce progeny at your facility, a new order and fee will be necessary.Cryorecovery to establish a Dedicated Supply for greater quantities of mice.
Mice recovered can be used to establish a dedicated colony to contractually supply you mice according to your requirements. Price by quotation. For more information on Dedicated Supply, please contact JAX® Services, Tel: 1-800-422-6423 (from U.S.A., Canada or Puerto Rico only) or 1-207-288-5845 (from any location).
|
|
Cryopreserved. Ready for recovery. Please refer to pricing and supply notes on the strain data sheet for further information.
| Control | ||
|---|---|---|
| Wild-type from the colony | ||
| 101045 B6129SF2/J | (approximate) | |
| Considerations for Choosing Controls | ||
| Control Pricing Information for Genetically Engineered Mutant Strains. | ||
For Licensing and Use Restrictions view the link(s) below:
- Use of MICE by companies or for-profit entities requires a license prior to shipping.
| phone: | 207-288-6470 |
| fax: | 207-288-6655 |
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