Rab29-dependent asymmetrical activation of leucine-rich repeat kinase 2.

Zhu, Hanwen; Tonelli, Francesca; Turk, Martin; Prescott, Alan; Alessi, Dario R; Sun, Ji · Science · 2023

basic_science · Level V

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Abstract

Gain-of-function mutations in <i>LRRK2</i>, which encodes the leucine-rich repeat kinase 2 (LRRK2), are the most common genetic cause of late-onset Parkinson's disease. LRRK2 is recruited to membrane organelles and activated by Rab29, a Rab guanosine triphosphatase encoded in the <i>PARK16</i> locus. We present cryo-electron microscopy structures of Rab29-LRRK2 complexes in three oligomeric states, providing key snapshots during LRRK2 recruitment and activation. Rab29 induces an unexpected tetrameric assembly of LRRK2, formed by two kinase-active central protomers and two kinase-inactive peripheral protomers. The central protomers resemble the active-like state trapped by the type I kinase inhibitor DNL201, a compound that underwent a phase 1 clinical trial. Our work reveals the structural mechanism of LRRK2 spatial regulation and provides insights into LRRK2 inhibitor design for Parkinson's disease treatment.

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