Skip to main page content
Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
Review
, 8, 420
eCollection

Modeling WWOX Loss of Function in vivo: What Have We Learned?

Affiliations
Review

Modeling WWOX Loss of Function in vivo: What Have We Learned?

Mayur Tanna et al. Front Oncol.

Abstract

The WW domain-containing oxidoreductase (WWOX) gene encompasses a common fragile sites (CFS) known as FRA16D, and is implicated in cancer. WWOX encodes a 46kDa adaptor protein, which contains two N-terminal WW-domains and a catalytic domain at its C-terminus homologous to short-chain dehydrogenase/reductase (SDR) family proteins. A high sequence conservation of WWOX orthologues from insects to rodents and ultimately humans suggest its significant role in physiology and homeostasis. Indeed, data obtained from several animal models including flies, fish, and rodents demonstrate WWOX in vivo requirement and that its deregulation results in severe pathological consequences including growth retardation, post-natal lethality, neuropathy, metabolic disorders, and tumorigenesis. Altogether, these findings set WWOX as an essential protein that is necessary to maintain normal cellular/physiological homeostasis. Here, we review and discuss lessons and outcomes learned from modeling loss of WWOX expression in vivo.

Keywords: Drosophila; cancer; epilepsy; knockout; metabolism; model organisms; mouse; zebrafish.

Figures

Figure 1
Figure 1
Phenotypes of WWOX deletion observed in different animal models.

Similar articles

See all similar articles

Cited by 4 articles

References

    1. Sutherland GR. Heritable fragile sites on human chromosomes I. Factors affecting expression in lymphocyte culture. Am J Hum Genet. (1979) 31:125–35. - PMC - PubMed
    1. Sutherland GR, Baker E, Seshadri RS. Heritable fragile sites on human chromosomes. V. A new class of fragile site requiring BrdU for expression. Am J Hum Genet. (1980) 32:542–8. - PMC - PubMed
    1. Sutherland GR, Jacky PB, Baker EG. Heritable fragile sites on human chromosomes. XI. Factors affecting expression of fragile sites at 10q25, 16q22, and 17p12. Am J Hum Genet. (1984) 36:110–22. - PMC - PubMed
    1. Glover TW, Berger C, Coyle J, Echo B. DNA polymerase alpha inhibition by aphidicolin induces gaps and breaks at common fragile sites in human chromosomes. Hum Genet. (1984) 67:136–42. 10.1007/BF00272988 - DOI - PubMed
    1. Durkin SG, Glover TW. Chromosome fragile sites. Annu Rev Genet. (2007) 41:169–92. 10.1146/annurev.genet.41.042007.165900 - DOI - PubMed

LinkOut - more resources

Feedback