Summary

RBM20 cardiomyopathy is a severe and arrhythmogenic form of DCM caused by pathogenic variants in the cardiac splicing factor RBM20. In FP-1, we investigated the role of the RBM20 target CAMK2D, and the role of LMNA phosphorylation in the development of the disease. We discovered that RBM20 cardiomyopathy is driven by excessive CAMK2D activation, and we could inhibit development and progression of disease by CAMK2D inhibition (Figure 1). Furthermore, even though we found no evidence of a critical role for LMNA phosphorylation in RBM20 cardiomyopathy, we did uncover a novel role of phosphorylated LMNA in RNA splicing. These results led to new questions, that will be addressed in FP-2, where we will merge with A06.

A05 graphic - Role of the splice target CAMK2D in RBM20 cardiomyopathy

SELECTED SCIENTIFIC OUTCOMES

2026

Pant P, Huang Y, Ghouse Z, Bai F, Kemmling E, Konrad L, Alameldeen A, Kistler R, Seeger T, Gotthardt M, Parikh VN, van den Hoogenhof MMG.

Circ Genom Precis Med. 19(4):e005471, 2026.

2026

van den Hoogenhof MMG, Duran J, Britto-Borges T, Sequeira V, Kemmling E, Konrad L, Schreiter F, Lennermann DC, Hartmann J, Schraft L, Kornienko J, Bock T, Krüger M, Maack C, Dieterich C, Steinmetz LM, Dewenter M, Backs J.

Nat Cardiovasc Res. 5(5):479-491, 2026.

2025

Kornienko, J., Schraft, L., Fenzl, K., van den Hoogenhof, MMG, Steinmetz, LM.

Circulation. 2025, 152(10), 739.

2025

van den Hoogenhof MM, Duran J, Britto-Borges T, Kemmling E, Konrad L, Lennermann DC, Hartmann J, Schraft L, Kornienko J, Bock T, Krüger M, Dieterich C, Steinmetz LM, Dewenter M & Backs J.

bioRxiv. 2025.05.23.655557, 2025.

2025

Pant P HY, Ghouse Z, Bai F, Kemmling E, Konrad L, Kistler R, Seeger T, Gotthardt M, Parikh VN & van den Hoogenhof MMG.

medRxiv. 2025.

2023

Grosch M, Schraft L, Chan A, Kuchenhoff L, Rapti K, Ferreira AM, Kornienko J, Li S, Radke MH, Kramer C, Clauder-Munster S, Perlas E, Backs J, Gotthardt M, Dieterich C, van den Hoogenhof MMG, Grimm D & Steinmetz LM.

Nat Commun. 14:3714, 2023.

2022

Lennermann DC, Pepin ME, Grosch M, Konrad L, Kemmling E, Hartmann J, Nolte JL, Clauder-Munster S, Kayvanpour E, Sedaghat-Hamedani F, Haas J, Meder B, van den Boogaard M, Amin AS, Dewenter M, Kruger M, Steinmetz LM, Backs J & van den Hoogenhof MMG.

Am J Physiol Heart Circ Physiol. 323:H1296-H310, 2022.

2021

Duran J, Nickel L, Estrada M, Backs J & van den Hoogenhof MMG.

Front Cell Dev Biol. 9:644630, 2021.

Scientific Staff

Zakiya Ghouse
Priyanka Pant

© Tobias Schwerdt, Max Delbrück Center