RT Journal Article SR Electronic T1 The Troyer syndrome protein spartin mediates selective autophagy of lipid droplets JF bioRxiv FD Cold Spring Harbor Laboratory SP 2021.08.18.456894 DO 10.1101/2021.08.18.456894 A1 Jeeyun Chung A1 Joongkyu Park A1 Zon Weng Lai A1 Talley J. Lambert A1 Ruth C. Richards A1 Robert V. Farese, Jr. A1 Tobias C. Walther YR 2021 UL http://biorxiv.org/content/early/2021/08/19/2021.08.18.456894.abstract AB Lipid droplets (LDs) are crucial organelles for energy storage and lipid homeostasis. Autophagy of LDs is an important pathway for their catabolism, but the molecular mechanisms mediating targeting of LDs for degradation by selective autophagy (lipophagy) are unknown. Here we identify spartin as a receptor localizing to LDs and interacting with core autophagy machinery, and we show that spartin is required to deliver LDs to lysosomes for triglyceride (TG) mobilization. Mutations in SPART (encoding spartin) lead to Troyer syndrome, a form of hereditary spastic paraplegia. We find that interfering with spartin function leads to LD and TG accumulation in motor cortex neurons of mice. Our findings thus identify spartin as a lipophagy receptor and suggest that impaired LD turnover may contribute to Troyer syndrome development.Competing Interest StatementThe authors have declared no competing interest.