Muskelin is a substrate adaptor of the highly regulated Drosophila embryonic CTLH E3 ligase

The maternal-to-zygotic transition (MZT) is a conserved developmental process where the maternally-derived protein and mRNA cache is replaced with newly made zygotic gene products. We have previously shown that in Drosophila the deposited RNA-binding proteins ME31B, Cup, and Trailer Hitch are ubiquitylated by the CTLH E3 ligase and cleared. However, the organization and regulation of the CTLH complex remain poorly understood in flies because Drosophila lacks an identifiable substrate adaptor, and the mechanisms restricting the degradation of ME31B and its cofactors to the MZT are unknown. Here, we show that the developmental regulation of the CTLH complex is multi-pronged, including transcriptional control by OVO and autoinhibition of the E3 ligase. One major regulatory target is the subunit Muskelin, which we demonstrate is a substrate adaptor for the Drosophila CTLH complex. Finally, we find that Muskelin has few targets beyond the three known RNA-binding proteins, showing exquisite target specificity. Thus, multiple levels of integrated regulation restrict the activity of the embryonic CTLH complex to early embryogenesis, during which time it regulates three important RNA-binding proteins.

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Publication Details

Journal
EMBO Reports
Published
2025-02-20
DOI
https://doi.org/10.1038/s44319-025-00397-6
Citations
10
Primary Topic
RNA Research and Splicing
Type
article
Field-Weighted Citation Impact
8.06

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article

Muskelin is a substrate adaptor of the highly regulated Drosophila embryonic CTLH E3 ligase

Addison B. Rains, Leif Benner, Philip R. Gafken, Chloe A. Briney et al.
10 citations
EMBO Reports
RNA Research and Splicing
8.06
article

Muskelin is a substrate adaptor of the highly regulated Drosophila embryonic CTLH E3 ligase

Addison B. Rains, Leif Benner, Philip R. Gafken, Chloe A. Briney, Chien-Chen Lin, Jesslyn C. Henriksen, Lisa Jones, Olivia S. Rissland, Roxana Gutierrez
article en
10 citations

Abstract

The maternal-to-zygotic transition (MZT) is a conserved developmental process where the maternally-derived protein and mRNA cache is replaced with newly made zygotic gene products. We have previously shown that in Drosophila the deposited RNA-binding proteins ME31B, Cup, and Trailer Hitch are ubiquitylated by the CTLH E3 ligase and cleared. However, the organization and regulation of the CTLH complex remain poorly understood in flies because Drosophila lacks an identifiable substrate adaptor, and the mechanisms restricting the degradation of ME31B and its cofactors to the MZT are unknown. Here, we show that the developmental regulation of the CTLH complex is multi-pronged, including transcriptional control by OVO and autoinhibition of the E3 ligase. One major regulatory target is the subunit Muskelin, which we demonstrate is a substrate adaptor for the Drosophila CTLH complex. Finally, we find that Muskelin has few targets beyond the three known RNA-binding proteins, showing exquisite target specificity. Thus, multiple levels of integrated regulation restrict the activity of the embryonic CTLH complex to early embryogenesis, during which time it regulates three important RNA-binding proteins.

EMBO ReportsVol. 26(6)
National Institutes of Health (US), Cape Town HVTN Immunology Laboratory / Hutchinson Centre Research Institute of South Africa (ZA), Fred Hutch Cancer Center (US), National Institute of Diabetes and Digestive and Kidney Diseases (US), University of Colorado Anschutz Medical Campus (US)
National Science Foundation, National Cancer Institute, National Institute of General Medical Sciences, National Institute of Diabetes and Digestive and Kidney Diseases, Office of Research Infrastructure Programs, National Institutes of Health
Openalex Percentile: Top 2%
RNA Research and Splicing
8.06
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