The PIDDosome controls cardiomyocyte polyploidization during postnatal heart development

The adult mammalian heart is characterized by post-mitotic polyploid cardiomyocytes (CMs). Understanding how CMs regulate cell cycle exit and polyploidy can help developing new heart regenerative therapies. Here, we uncover that the PIDDosome, a multi-protein complex activating the endopeptidase Caspase-2, helps to implement a CM-specific differentiation program that limits ploidy during postnatal heart development. DNA content analyses show that cell-autonomous PIDDosome loss causes an increase in nuclear and cellular CM ploidy. Increased ploidy does not affect cardiac structure nor function in early adulthood, but correlates with a modest reduction in cardiac performance in aged mice. PIDDosome-imposed polyploidy control commences at postnatal day 7 (P7), reaching a plateau by P14. PIDDosome activation requires ANKRD26, targeting PIDD1 to mother centrioles. Opposite to prior observations in liver development, the PIDDosome limits CM polyploidization in a p53-independent manner but reliant on induction of p21/Cdkn1a, a notion supported by nuclear RNA sequencing and genetic deletion experiments. Our results provide new insights how proliferation of polyploid CMs is restricted during postnatal heart development.

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Publication Details

Journal
Cell Death and Differentiation
Published
2026-01-12
DOI
https://doi.org/10.1038/s41418-025-01645-x
Citations
1
Primary Topic
Congenital heart defects research
Type
article
Field-Weighted Citation Impact
22.24

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article

The PIDDosome controls cardiomyocyte polyploidization during postnatal heart development

R. Hirschberger, D. Obwegs, N. Kinz, V. C. Sladky et al.
1 citations
Cell Death and Differentiation
Congenital heart defects research
22.24
article

The PIDDosome controls cardiomyocyte polyploidization during postnatal heart development

R. Hirschberger, D. Obwegs, N. Kinz, V. C. Sladky, M. Leone, P. Giansanti, R. Yousif, M. A. Sussman, F. Eichin, M. Boerries, J. Mergner, M. M. Marques, L. Englmaier, L. S. Maupome, N. Martinez-Garcia, K. Moos, L. Eblahed, D. Rizzotto, E. D. Jacotot, D. Dawood, V. Z. Braun, M. K. Wright, C. Savko, C. Manzl, A. Villunger
article en
1 citations

Abstract

The adult mammalian heart is characterized by post-mitotic polyploid cardiomyocytes (CMs). Understanding how CMs regulate cell cycle exit and polyploidy can help developing new heart regenerative therapies. Here, we uncover that the PIDDosome, a multi-protein complex activating the endopeptidase Caspase-2, helps to implement a CM-specific differentiation program that limits ploidy during postnatal heart development. DNA content analyses show that cell-autonomous PIDDosome loss causes an increase in nuclear and cellular CM ploidy. Increased ploidy does not affect cardiac structure nor function in early adulthood, but correlates with a modest reduction in cardiac performance in aged mice. PIDDosome-imposed polyploidy control commences at postnatal day 7 (P7), reaching a plateau by P14. PIDDosome activation requires ANKRD26, targeting PIDD1 to mother centrioles. Opposite to prior observations in liver development, the PIDDosome limits CM polyploidization in a p53-independent manner but reliant on induction of p21/Cdkn1a, a notion supported by nuclear RNA sequencing and genetic deletion experiments. Our results provide new insights how proliferation of polyploid CMs is restricted during postnatal heart development.

Cell Death and Differentiation
Austrian Academy of Sciences (AT), Innsbruck Medical University (AT), Inserm (FR), University of Freiburg (DE), Université Paris Cité (FR), San Diego State University (US), University Medical Center Freiburg (DE), TUM Klinikum (DE), Sorbonne Paris Cité (FR), CeMM Research Center for Molecular Medicine (AT), Laboratoire de Recherche Vasculaire Translationnelle (FR), Technical University of Munich (DE), Universidad de León (ES)
Johns Hopkins University
Good health and well-being
Openalex Percentile: Top 3%
Congenital heart defects research
22.24
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