BORC assemblies integrate BLOC-1 subunits to diversify endosomal trafficking functions

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Authors: Mariana E. G. de Araujo, Sascha Josef Amann, Taras Stasyk, Alexander Schleiffer, Eva Rauch, Paula Flümann, Isabel I. Singer, Leopold Kremser, Vojtěch Dostál, Thanida Laopanupong, Nikolaus Obojes, Moritz H. Wallnöfer
+11 more Flora S. Gradl, R Kurzbauer, Caroline Krebiehl, Samuel Kofler, Irina Grishkovskaya, Georg Friedrich Vogel, Michael W. Hess, Bettina Sarg, Tim Clausen, David Haselbach, Lukas Alfons Huber
Cited by: 1
References: 52

Abstract

BORC and BLOC-1 are multisubunit complexes that regulate endolysosomal trafficking. Although they are presumed to be distinct, their paralogous origins and shared subunits suggest the potential for higher-order assembly. Here, we reveal the conserved octameric architecture of BORC formed by two intertwined tetramers and present the structure of C. elegans BORC. Through cross-linking mass spectrometry of endogenous complexes, we validate this model for human BORC and demonstrate that the integrity of the complex, which is essential for lysosomal transport, relies on specific interfacial residues. We also clarify the disruptive nature of disease-causing mutations and propose that the formation and function of BORC are likely regulated by specific cues. These cues might include the phosphorylation of Snapin and a pH-sensitive histidine residue in BORCS5. Additionally, we present direct biochemical and structural evidence of BORC-BLOC-1 hybrid complexes. Finally, we link a specific hybrid complex to the regulation of transferrin receptor recycling via interaction with the EARP complex. Our work challenges the paradigm of BORC and BLOC-1 as separate entities, establishing a model of dynamic complex formation wherein modular assembly creates functional specialization to meet diverse cellular demands.

Fields of Study & Subjects

Cellular transport and secretion Lysosomal Storage Disorders Research Calcium signaling and nucleotide metabolism

Publication details

Journal / source
Proceedings of the National Academy of Sciences
Publisher
National Academy of Sciences
Volume
123
Issue
4
Pages
e2515691123–e2515691123
Publication date
20 January 2026
Language
EN