Click here to see zebrafish mutants.
======================================================================================== | | Mutant type | Phenotype | Reference (1st author) | ======================================================================================== | Angiotensin | transgenic | cortical thinning and | Krege 1995 | | converting | | focal atrophy in | | enzyme | | adulthood | --------------------------------------------------------------------------------------- | Angiotensinogen | transgenic | vessel walls thicken | Kim 1995 | | | | during post-natal | | | | development | --------------------------------------------------------------------------------------- | AP-2 | transgenic | hypoplastic, retarded | Zhang 1996 | | | | development of meta- | | | | nephros (& many other | | | | defects in embryo) | --------------------------------------------------------------------------------------- | Bcl-2 -/- | transgenic | polycystic disease | Veis 1993 | | | | hypoplasia due to | Nagata 1996 | | | excess apoptosis | | | | during abnormal | | | | neprhogenesis | --------------------------------------------------------------------------------------- | BF-2 | transgenic | hypoplastic, fused, | Hatini 1996 | | | | c.d. small, too few | | | | nephrons,large mesen- | | | | chymal condenates | --------------------------------------------------------------------------------------- | BMP4 +/- | transgenic | polycystic | Dunn 1997 | | | | during post-natal | | | | development | --------------------------------------------------------------------------------------- | BMP-7 | transgenic-1|1 severe kid hypoplasia| Dudley 1995 | | | (lethal): early devt | Luo 1995 | | | OK (->E12.5), but then| | | | few nephrons form and | | | | cd branching ceases | | | | (according to Dudley &| | | | Luo papers). | | | transgenic-2|2 adequate glomerulo- | Jena 1997 | | | genesis, well-formed | | | | proximal tubule but | | | | poor distal tubules, | | | | polycystic. | --------------------------------------------------------------------------------------- | Collagen II | transgenic | minor defects | Li 1995 | | alpha 1 | | | | | | | --------------------------------------------------------------------------------------- | Collagen IV | transgenic | Alport's syndrome | Cosgrove 1996 | | alpha 3 | | | --------------------------------------------------------------------------------------- | Cox-2 | transgenic | poor bud development | Dinchuk 1995 | (cyclooxygenase) | | apparent by birth | | | | nephrons few and | | | | immature | | | | (E14 kidneys look OK | ---------------------------------------------------------------------------------------- | Emx-2 | transgenic | absence of metanephroi| Pellegrini 1996 | --------------------------------------------------------------------------------------- | Epstein-Barr | transgenic | tubular hyperplasia | Tornell 1996 | | nuclear Antigen 2| | then neoplasia | | ---------------------------------------------------------------------------------------- | Fu (Fused) | random | variable failure of | Theiler 1956 | | | | ureteric bud growth | | ---------------------------------------------------------------------------------------- | FGF-7 (=KGF) | transgenic | hyperplastic, cystic | Nguyen et al 1996 | | overexpression | | renal collecting | | | targeted to | | duct system. | | | liver (not kid) | | | | ---------------------------------------------------------------------------------------- | FGF-7 knockout | transgenic | kidneys smaller than | Qiao et al 1999 | | | | +/+ wrt both coll | | | | | duct system size and | | | | | neprhon number | | ---------------------------------------------------------------------------------------- | GDNF | transgenic | renal agenesis due | Sanchez et al 1996 | | | | to lack of ureteric | Pichel et al 1996 | | | | bud. Can be rescued in| Moore et al 1996 | | | | vivo by added GDNF | Sainio et al 1997 | ---------------------------------------------------------------------------------------- | Glucose-6-phosph-| transgenic | tubules enlarge at the| Lei 1996 | | atase | | expense of glomeruli | --------------------------------------------------------------------------------------- | Heparan sulphate | transgenic | no branching of ub, | Bullock 1998 | | 2-sulphotransfer-| | no condensation of | | ase | | mesenchyme | --------------------------------------------------------------------------------------- | Hexosaminidase | transgenic | vacuolation | Phaneuf 1996 | | | | during post-natal | | | | development | --------------------------------------------------------------------------------------- | HNF-1 | transgenic | Fanconi syndrome | Pontoglio 1996 | | | | during post-natal | | | | development | --------------------------------------------------------------------------------------- | Hox a-11 + | transgenic | severe kidney defects | Davis et al 1995 | | Hox d-11 | | | | | double | | | | | mutant | | | | ---------------------------------------------------------------------------------------- | IGF I -/- | transgenic | hypoplasia in line | Liu et al 1993 | | | | with that of the whole| | | | | animal (no SPECIFIC | | | | | renal effects) | | ---------------------------------------------------------------------------------------- | IGFIR -/- | transgenic | similar to IGFI | Liu et al 1993 | ---------------------------------------------------------------------------------------- | IGFII -/- | transgenic | similar to IGF I above| DeChiara et al 1990 | ---------------------------------------------------------------------------------------- | integrin | transgenic | too few collecting | Kreidberg 1996 | | alpha-3 -/- | | duct branches; cystic | | | | | proximal tubules | | ---------------------------------------------------------------------------------------- | integrin | transgenic | failure of ureteric | Muller 1997 | | alpha-8 -/- | | bud growth; nephrons | | | | | don't epithelialize | | ---------------------------------------------------------------------------------------- | ld (limb | random | variable failure of | Maas 1994 | | deformity - | | ureteric bud growth | | | affects formins | | | | ---------------------------------------------------------------------------------------- | LIM-1 -/- | transgenic | failure of kidney and | Swawbrot and Behringer 1995 | | | | genital development | | ---------------------------------------------------------------------------------------- | Neuraminidase | transgenic | cell vacuolation | Zhou 1995 | --------------------------------------------------------------------------------------- |Os/+ | random | hypoplasia in vivo and| Sorenson et al 1996 | | (oligosyndactyly)| | in culture | | ---------------------------------------------------------------------------------------- | p53 over- | transgenic | Hypoplastic; defective| Godley et al. 1996 | | expression | | collecting duct diffn.| | | | High apoptosis of | | | | nephrogenic mesenchyme| | | | 'inefficient convers- | | | | ion to tubules' | ---------------------------------------------------------------------------------------- | p57-KIP2 -/- | transgenic | medullary dysplasia | Zhang et al. 1997 | | | | like Beckwith- | | | | Wiedemann syndrome | ---------------------------------------------------------------------------------------| | Pax-2 -/- | transgenic | failure of mesonephros| Torres et al. 1995 | | | | metanephros & genital | | | | development. Neither | | | | tubules nor ureteric | | | | bud form. Even hetero-| | | | zygote has small kid's| ---------------------------------------------------------------------------------------| | PDGF-B -/- | transgenic | very few glomeruli | Leveen 1995 |
| | | | Lindahl 1998 |
---------------------------------------------------------------------------------------- | PDGF-R -/- | transgenic | very few glomeruli | Soriano 1994 |
| | | | Lindahl 1998 | ---------------------------------------------------------------------------------------- | Retinoic acid | transgenic | aplasia (due to lack | Moens 1993 | | receptor -/- | | of ureteric bud) & | | | (but see table | | hypoplasia. Variable | | | below and | | phenotype; some RAR | | | entry below) | | subtype redundancy | | ---------------------------------------------------------------------------------------- | Retinoic acid | transgenic | Impaired stromal | Mendelsohn 1999 | | receptor (RAR) | | function, poor ub | | | alpha / beta 1 | | branching. c-Ret down-| | | double KO | | regulated. | | ---------------------------------------------------------------------------------------- | c-ret -/- | transgenic | variable failure of | Pachnis 1993 | | | | ureteric bud invasion,| Schuchardt 1994 | | | | growth and branching. | | | | | MM can still be induced | | | | by spinal cord. | | ---------------------------------------------------------------------------------------- | c-ret driven | transgenic | variable failure of | Srinivas 1999 | | by hox b7 | | ureteric bud | | | promoter | | growth and branching. | | | (= expression | | (see paper - this | | | throughout bud)| | summary too brief) | | ---------------------------------------------------------------------------------------- | Sd (Danforth's | random | hypoplastic/aplastic | Gluecksohn-Schoenheimer 1943| | short tail) | | kidneys (ureteric bud | | | | | invasion inhibited) | | ---------------------------------------------------------------------------------------| | Shh (Somic | transgenic | Kidney defect of | Chiang 1996 | hedgehog | | unspecified type | | | | | | | ---------------------------------------------------------------------------------------| | Uteroglobin | transgenic | FN-deposit glomerular | Zhang 1997 | knockout | | disease | | | | | | | ---------------------------------------------------------------------------------------| | Wg (chicken | random | Failure of ureteric | Secondary source: Ede 1978 -| | wingless) | | bud invasion | anyone know the primary | | (presumably | | | source? (please mail me) | | nothing to do | | | | | with Drosophila | | | | | Wg or Wnt genes) | | | | ---------------------------------------------------------------------------------------- | Wnt4 -/- | transgenic | MM won't epithelialise| Stark 1994 | | | | and wnt4 transcription| | | | | not activated (not even | | | | the mutant message) | | ---------------------------------------------------------------------------------------- | WT-1 -/- | transgenic | absence of metanephro-| Kreidberg 1993 | | | | genic mesenchyme | | ----------------------------------------------------------------------------------------
--------------------------------------------------------------------------- | Mutant | type | Reference | =========================================================================== | BMP-5 | random (se) | King 1994 | | CRABPI | transgenic | de Bruijn 1994 | | HGF | transgenic | Birchmeier pers comm | | Hox C9 | transgenic | Suemori 1995 | | Hox 11 (alone) | transgenic | Hseih-Li 1995 | | Integrin alpha 6 | transgenic | Georges-Labouesse 1996 | LIFR | transgenic | Ware et al 1995 | | MMP-9 -/- | transgenic | Andrews et al 2000 |
| NCAM | transgenic | Cramer 94 | | p75 NGFR | transgenic | Lee et al. 1992 | | RAR betas (all) | transgenic | Luo et al. 1995 (but see above) | RAR beta-2 | transgenic | Mendelsohn 1994b (but see above) | | Vimentin | transgenic | Collucci-Guyon 94 | | Wnt1 | transgenic | Wilkinson 87 | ---------------------------------------------------------------------------