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Chd7 regulates lipid metabolism and swim bladder inflation in zebrafish

Breuer, Maximillian, Fortier, Gabrielle, Jamadagni, Priyanka, Zaouter, Charlotte, Kassa, Betelhem et Patten, Shunmoogum A. . Chd7 regulates lipid metabolism and swim bladder inflation in zebrafish In: 11e European Zebrafish Meeting, 26-27 October 2020, virtuel.

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Résumé

The chromodomain helicase remodelling enzyme, CHD7, is a key regulator of several pathways from neural crest cell differentiation to stem cell maintenance. Mutations in CHD7 have been directly linked to CHARGE syndrome- a syndrome characterized by multisystemic symptoms, including craniofacial dysmorphisms and heart defects. Our study shows that chd7-/- zebrafish present with characteristics of a dyslipidemia. Specifically, we identify accumulation of neutral lipid droplets in the swim bladder and vascular system of developing larvae. Along these lines we identified a collection of dysregulated lipid metabolism enzymes along with pparγ regulated lipid metabolism. Following, we could link lipid metabolism to swim bladder development and observe decreased size and non-inflated swim bladder during larval development dependent on proper lipid metabolism in the surfactant system. In detail we could show that loss of elongation factor 1, elovl1, and the fatty acid binding protein 7b, fabp7b, are dependent for the lipid metabolism of the swim bladder and subsequently lead to failure to inflate the swim bladder. Our study is the first to investigate the multisystemic role of CHD7 in correlation to lipid metabolism and swim bladder development and its fundamental underlying pathway.

Type de document: Document issu d'une conférence ou d'un atelier
Informations complémentaires: Affiche scientifique P042
Mots-clés libres: -
Centre: Centre INRS-Institut Armand Frappier
Date de dépôt: 14 juill. 2021 15:41
Dernière modification: 14 juill. 2021 15:41
URI: https://espace.inrs.ca/id/eprint/11855

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