The zebrafish cohesin protein Sgo1 is required for cardiac function and eye development

Sarah M Kamel, Sanne Broekman, Federico Tessadori, Erwin van Wijk, Jeroen Bakkers

Onderzoeksoutput: Bijdrage aan wetenschappelijk tijdschrift/periodieke uitgaveArtikelWetenschappelijkpeer review

1 Citaat (Scopus)

Samenvatting

BACKGROUND: Cohesinopathies is a term that refers to/covers rare genetic diseases caused by mutations in the cohesin complex proteins. The cohesin complex is a multiprotein complex that facilitates different aspects of cell division, gene transcription, DNA damage repair, and chromosome architecture. Shugoshin proteins prevent the cohesin complex from premature dissociation from chromatids during cell division. Patients with a homozygous missense mutation in SGO1, which encodes for Shugoshin1, have problems with normal pacing of the heart and gut.

RESULTS: To study the role of shugoshin during embryo development, we mutated the zebrafish sgo1 gene. Homozygous sgo1 mutant embryos display various phenotypes related to different organs, including a reduced heart rate accompanied by reduced cardiac function. In addition, sgo1 mutants are vision-impaired as a consequence of structurally defective and partially non-functional photoreceptor cells. Furthermore, the sgo1 mutants display reduced food intake and early lethality.

CONCLUSION: We have generated a zebrafish model of Sgo1 that showed its importance during organ development and function.

Originele taal-2Engels
Pagina's (van-tot)1357-1367
Aantal pagina's11
TijdschriftDevelopmental Dynamics
Volume251
Nummer van het tijdschrift8
DOI's
StatusGepubliceerd - aug. 2022

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