Poster Presentation 40th Annual Lorne Genome Conference 2019

Elk1 in Congenital and Late Onset Cardiac Disease: at the Heart of the Matter (#164)

Jeannette JCH Hallab 1 , Gonzalo GDM Del Monte-Nieto 2 , Inken IH Huttner 2 , Akriti AV Varshney 1 , Celine CS Santiago 2 , Romaric RB Bouveret 3 , Daniel DH Hesselson 4 , Lee LM Miles 5 , Sebastian SD Dworkin 5 , Kazu KK Kikuchi 2 , Diane DF Fatkin 2 , Richard RH Harvey 2 , Mirana MR Ramialison 1
  1. Australian Regenerative Medicine Institute, Monash Uni, Clayton, VIC, Australia
  2. The Victor Chang Cardiac Research Institute, Sydney
  3. The University of New South Wales, Sydney
  4. The Garvan Insitute for Medical Research, Sydney, NSW
  5. La Trobe University, Melbourne

Elk1 is an ETS Class I, TCF subfamily transcription factor known as a well- established downstream effector

of the MAPK pathway and has been implicated in the causation of a variety of cancers. Recent in-vitro

evidence places Elk1 in the context of the cardiogenic transcription factor network, although its in-vivo role

in cardiogenesis remains unexplored.

We provide the first in-vivo evidence of the role of Elk1 in cardiogenesis using a zebrafish mutant with

disrupted DNA binding domain (elk1 -543/-543) and cardiac defects including valve displacement and

elongation with hypertrophic/hyperplastic changes in the ventricular myocardium. elk1 -543/-543 are

predisposed to early embryonic death, with high incidence of heart looping defects and accelerated growth

among survivors.

RNA-seq at 6dpf provides insights into the basis of heart defects, indicating up-regulation of MAPK pathway

genes and down-regulation of trim63a, encoding a homeostatic protein involved in reducing muscle mass,

dys-regulation of which is associated with Hypertrophic Cardiomyopathy (HCM) in humans.

MAPK pathway up-regulation is commonly associated with HCM although the fundamental basis of this

relationship is not completely understood. We provide mechanistic insight, suggesting MAPK perturbations

could converge via the TCFs at a (putative) trim63a enhancer, down-regulating trim63a to mediate HCM.

Early developmental RNA-seq indicates loss of Elk1 function is associated with down-regulation of tumor

suppressor genes. We hypothesize this promotes embryonic survival via non-optimal pathways, and

fundamentally underlies observed defects.

The sum of changes in elk1 -543/-543 mimic a group of congenital syndromes known as “RASopathies" in

humans. Our data provides important insights into the time line of molecular events underlying

RASopathies/MAPK pathway defects and their relationship to molecules imperative in heart patterning and

homeostasis.