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.
Authors
- 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
Institutions
- 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)
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
Funders
- Johns Hopkins University