Zadoorian A, Du X, Yang H. Lipid droplet biogenesis and functions in health and disease. Nat Rev Endocrinol. 2023;19(8):443–59.

Article  PubMed  CAS  Google Scholar 

Rakotonirina-Ricquebourg R, Costa V, Teixeira V. Hello from the other side: Membrane contact of lipid droplets with other organelles and subsequent functional implications. Prog Lipid Res. 2022;85:101141.

Article  PubMed  CAS  Google Scholar 

Henne W. The Molecular Era of Lipid Droplets. Contact. 2020;3:1–9.

Yan R, Qian H, Lukmantara I, Gao M, Du X, Yan N, Yang H. Human SEIPIN Binds Anionic Phospholipids. Dev Cell. 2018;47(2):248-56 e4.

Article  PubMed  CAS  Google Scholar 

Combot Y, Salo VT, Chadeuf G, Holtta M, Ven K, Pulli I, et al. Seipin localizes at endoplasmic-reticulum-mitochondria contact sites to control mitochondrial calcium import and metabolism in adipocytes. Cell Rep. 2022;38(2):110213.

Article  PubMed  CAS  Google Scholar 

Fei W, Shui G, Gaeta B, Du X, Kuerschner L, Li P, et al. Fld1p, a functional homologue of human seipin, regulates the size of lipid droplets in yeast. J Cell Biol. 2008;180(3):473–82.

Article  PubMed  PubMed Central  CAS  Google Scholar 

Klug YA, Deme JC, Corey RA, Renne MF, Stansfeld PJ, Lea SM, Carvalho P. Mechanism of lipid droplet formation by the yeast Sei1/Ldb16 Seipin complex. Nat Commun. 2021;12(1):5892.

Article  PubMed  PubMed Central  CAS  Google Scholar 

Arlt H, Sui X, Folger B, Adams C, Chen X, Remme R, et al. Seipin forms a flexible cage at lipid droplet formation sites. Nat Struct Mol Biol. 2022;29(3):194–202.

Article  PubMed  PubMed Central  CAS  Google Scholar 

Binns D, Lee S, Hilton CL, Jiang QX, Goodman JM. Seipin is a discrete homooligomer. Biochemistry. 2010;49(50):10747–55.

Article  PubMed  CAS  Google Scholar 

Grippa A, Buxo L, Mora G, Funaya C, Idrissi FZ, Mancuso F, et al. The seipin complex Fld1/Ldb16 stabilizes ER-lipid droplet contact sites. J Cell Biol. 2015;211(4):829–44.

Article  PubMed  PubMed Central  CAS  Google Scholar 

Fei W, Shui G, Zhang Y, Krahmer N, Ferguson C, Kapterian TS, et al. A role for phosphatidic acid in the formation of “supersized” lipid droplets. PLoS Genet. 2011;7(7):e1002201.

Article  PubMed  PubMed Central  CAS  Google Scholar 

Salo VT, Belevich I, Li S, Karhinen L, Vihinen H, Vigouroux C, et al. Seipin regulates ER-lipid droplet contacts and cargo delivery. EMBO J. 2016;35(24):2699–716.

Article  PubMed  PubMed Central  CAS  Google Scholar 

Szymanski KM, Binns D, Bartz R, Grishin NV, Li WP, Agarwal AK, et al. The lipodystrophy protein seipin is found at endoplasmic reticulum lipid droplet junctions and is important for droplet morphology. Proc Natl Acad Sci U S A. 2007;104(52):20890–5.

Article  PubMed  PubMed Central  CAS  Google Scholar 

Ito D, Fujisawa T, Iida H, Suzuki N. Characterization of seipin/BSCL2, a protein associated with spastic paraplegia 17. Neurobiol Dis. 2008;31(2):266–77.

Article  PubMed  CAS  Google Scholar 

Windpassinger C, Auer-Grumbach M, Irobi J, Patel H, Petek E, Horl G, et al. Heterozygous missense mutations in BSCL2 are associated with distal hereditary motor neuropathy and Silver syndrome. Nat Genet. 2004;36(3):271–6.

Article  PubMed  CAS  Google Scholar 

Auer-Grumbach M, Schlotter-Weigel B, Lochmuller H, Strobl-Wildemann G, Auer-Grumbach P, Fischer R, et al. Phenotypes of the N88S Berardinelli-Seip congenital lipodystrophy 2 mutation. Ann Neurol. 2005;57(3):415–24.

Article  PubMed  CAS  Google Scholar 

Teixeira V, Maciel P, Costa V. Leading the way in the nervous system: Lipid Droplets as new players in health and disease. Biochim Biophys Acta Mol Cell Biol Lipids. 2021;1866(1):158820.

Article  PubMed  CAS  Google Scholar 

van de Warrenburg BP, Scheffer H, van Eijk JJ, Versteeg MH, Kremer H, Zwarts MJ, et al. BSCL2 mutations in two Dutch families with overlapping Silver syndrome-distal hereditary motor neuropathy. Neuromuscul Disord. 2006;16(2):122–5.

Article  PubMed  Google Scholar 

Ito D, Yagi T, Suzuki N. BSCL2-related neurologic disorders/seipinopathy: endoplasmic reticulum stress in neurodegeneration. Rinsho Shinkeigaku. 2011;51(11):1186–8.

Article  PubMed  Google Scholar 

Ito D, Yagi T, Ikawa M, Suzuki N. Characterization of inclusion bodies with cytoprotective properties formed by seipinopathy-linked mutant seipin. Hum Mol Genet. 2012;21(3):635–46.

Article  PubMed  CAS  Google Scholar 

Yagi T, Ito D, Nihei Y, Ishihara T, Suzuki N. N88S seipin mutant transgenic mice develop features of seipinopathy/BSCL2-related motor neuron disease via endoplasmic reticulum stress. Hum Mol Genet. 2011;20(19):3831–40.

Article  PubMed  CAS  Google Scholar 

Ito D, Suzuki N. Molecular pathogenesis of seipin/BSCL2-related motor neuron diseases. Ann Neurol. 2007;61(3):237–50.

Article  PubMed  CAS  Google Scholar 

Carman GM, Han GS. Regulation of phospholipid synthesis in the yeast Saccharomyces cerevisiae. Annu Rev Biochem. 2011;80:859–83.

Article  PubMed  PubMed Central  CAS  Google Scholar 

Santiago TC, Mamoun CB. Genome expression analysis in yeast reveals novel transcriptional regulation by inositol and choline and new regulatory functions for Opi1p, Ino2p, and Ino4p. J Biol Chem. 2003;278(40):38723–30.

Article  PubMed  CAS  Google Scholar 

Lopes JM, Henry SA. Interaction of trans and cis regulatory elements in the INO1 promoter of Saccharomyces cerevisiae. Nucleic Acids Res. 1991;19(14):3987–94.

Article  PubMed  PubMed Central  CAS  Google Scholar 

Loewen CJ, Gaspar ML, Jesch SA, Delon C, Ktistakis NT, Henry SA, Levine TP. Phospholipid metabolism regulated by a transcription factor sensing phosphatidic acid. Science. 2004;304(5677):1644–7.

Article  PubMed  CAS  Google Scholar 

Jesch SA, Zhao X, Wells MT, Henry SA. Genome-wide analysis reveals inositol, not choline, as the major effector of Ino2p-Ino4p and unfolded protein response target gene expression in yeast. J Biol Chem. 2005;280(10):9106–18.

Article  PubMed  CAS  Google Scholar 

Henry SA, Gaspar ML, Jesch SA. The response to inositol: regulation of glycerolipid metabolism and stress response signaling in yeast. Chem Phys Lipids. 2014;180:23–43.

Article  PubMed  CAS  Google Scholar 

Loewen CJ, Levine TP. A highly conserved binding site in vesicle-associated membrane protein-associated protein (VAP) for the FFAT motif of lipid-binding proteins. J Biol Chem. 2005;280(14):14097–104.

Article  PubMed  CAS  Google Scholar 

Hofbauer HF, Gecht M, Fischer SC, Seybert A, Frangakis AS, Stelzer EHK, et al. The molecular recognition of phosphatidic acid by an amphipathic helix in Opi1. J Cell Biol. 2018;217(9):3109–26.

Article  PubMed  PubMed Central  CAS  Google Scholar