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

TY - JOUR

T1 - The zebrafish cohesin protein Sgo1 is required for cardiac function and eye development

AU - Kamel, Sarah M

AU - Broekman, Sanne

AU - Tessadori, Federico

AU - van Wijk, Erwin

AU - Bakkers, Jeroen

N1 - © 2022 The Authors. Developmental Dynamics published by Wiley Periodicals LLC on behalf of American Association for Anatomy.

PY - 2022/8

Y1 - 2022/8

N2 - 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.

AB - 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.

KW - Animals

KW - Cell Cycle Proteins/physiology

KW - Centromere/metabolism

KW - Chromatids/metabolism

KW - Chromosomal Proteins, Non-Histone/genetics

KW - Zebrafish/genetics

U2 - 10.1002/dvdy.468

DO - 10.1002/dvdy.468

M3 - Article

C2 - 35275424

SN - 1058-8388

VL - 251

SP - 1357

EP - 1367

JO - Developmental Dynamics

JF - Developmental Dynamics

IS - 8

ER -