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Walter Neupert (1939-2019), a pioneer of mitochondrial biogenesis and morphology.

Lill R, Langer T, Herrmann JM.

EMBO J. 2019 Aug 16:e103100. doi: 10.15252/embj.2019103100. [Epub ahead of print] No abstract available.


Depletion of thiol reducing capacity impairs cytosolic but not mitochondrial iron-sulfur protein assembly machineries.

Braymer JJ, Stümpfig M, Thelen S, Mühlenhoff U, Lill R.

Biochim Biophys Acta Mol Cell Res. 2019 Feb;1866(2):240-251. doi: 10.1016/j.bbamcr.2018.11.003. Epub 2018 Nov 10.


Branched late-steps of the cytosolic iron-sulphur cluster assembly machinery of Trypanosoma brucei.

Tonini ML, Peña-Diaz P, Haindrich AC, Basu S, Kriegová E, Pierik AJ, Lill R, MacNeill SA, Smith TK, Lukeš J.

PLoS Pathog. 2018 Oct 22;14(10):e1007326. doi: 10.1371/journal.ppat.1007326. eCollection 2018 Oct.


Function and crystal structure of the dimeric P-loop ATPase CFD1 coordinating an exposed [4Fe-4S] cluster for transfer to apoproteins.

Stehling O, Jeoung JH, Freibert SA, Paul VD, Bänfer S, Niggemeyer B, Rösser R, Dobbek H, Lill R.

Proc Natl Acad Sci U S A. 2018 Sep 25;115(39):E9085-E9094. doi: 10.1073/pnas.1807762115. Epub 2018 Sep 10.


ISCA1 mutation in a patient with infantile-onset leukodystrophy causes defects in mitochondrial [4Fe-4S] proteins.

Torraco A, Stehling O, Stümpfig C, Rösser R, De Rasmo D, Fiermonte G, Verrigni D, Rizza T, Vozza A, Di Nottia M, Diodato D, Martinelli D, Piemonte F, Dionisi-Vici C, Bertini E, Lill R, Carrozzo R.

Hum Mol Genet. 2018 Oct 15;27(20):3650. doi: 10.1093/hmg/ddy273. No abstract available.


A novel complex neurological phenotype due to a homozygous mutation in FDX2.

Gurgel-Giannetti J, Lynch DS, Paiva ARB, Lucato LT, Yamamoto G, Thomsen C, Basu S, Freua F, Giannetti AV, de Assis BDR, Ribeiro MDO, Barcelos I, Sayão Souza K, Monti F, Melo US, Amorim S, Silva LGL, Macedo-Souza LI, Vianna-Morgante AM, Hirano M, Van der Knaap MS, Lill R, Vainzof M, Oldfors A, Houlden H, Kok F.

Brain. 2018 Aug 1;141(8):2289-2298. doi: 10.1093/brain/awy172.


Conserved functions of Arabidopsis mitochondrial late-acting maturation factors in the trafficking of iron‑sulfur clusters.

Uzarska MA, Przybyla-Toscano J, Spantgar F, Zannini F, Lill R, Mühlenhoff U, Rouhier N.

Biochim Biophys Acta Mol Cell Res. 2018 Sep;1865(9):1250-1259. doi: 10.1016/j.bbamcr.2018.06.003. Epub 2018 Jun 12.


Fe-S cluster coordination of the chromokinesin KIF4A alters its subcellular localization during mitosis.

Ben-Shimon L, Paul VD, David-Kadoch G, Volpe M, Stümpfig M, Bill E, Mühlenhoff U, Lill R, Ben-Aroya S.

J Cell Sci. 2018 Jun 27;131(12). pii: jcs211433. doi: 10.1242/jcs.211433.


ISCA1 Mutation In A Patient With Infantile-Onset Leukodystrophy Causes Defects In Mitochondrial [4Fe-4S] Proteins.

Torraco A, Stehling O, Stümpfig C, Rösser R, De Rasmo D, Fiermonte G, Verrigni D, Rizza T, Vozza A, Di Nottia M, Diodato D, Martinelli D, Piemonte F, Dionisi-Vici C, Bertini E, Lill R, Carrozzo R.

Hum Mol Genet. 2018 May 14. doi: 10.1093/hmg/ddy183. [Epub ahead of print]


Biochemical Analyses of Human Iron-Sulfur Protein Biogenesis and of Related Diseases.

Stehling O, Paul VD, Bergmann J, Basu S, Lill R.

Methods Enzymol. 2018;599:227-263. doi: 10.1016/bs.mie.2017.11.004. Epub 2017 Dec 6.


Biochemical Reconstitution and Spectroscopic Analysis of Iron-Sulfur Proteins.

Freibert SA, Weiler BD, Bill E, Pierik AJ, Mühlenhoff U, Lill R.

Methods Enzymol. 2018;599:197-226. doi: 10.1016/bs.mie.2017.11.034. Epub 2018 Jan 10.


Human CIA2A-FAM96A and CIA2B-FAM96B Integrate Iron Homeostasis and Maturation of Different Subsets of Cytosolic-Nuclear Iron-Sulfur Proteins.

Stehling O, Mascarenhas J, Vashisht AA, Sheftel AD, Niggemeyer B, Rösser R, Pierik AJ, Wohlschlegel JA, Lill R.

Cell Metab. 2018 Jan 9;27(1):263. doi: 10.1016/j.cmet.2017.12.009. No abstract available.


The heme synthesis defect of mutants impaired in mitochondrial iron-sulfur protein biogenesis is caused by reversible inhibition of ferrochelatase.

Lange H, Mühlenhoff U, Denzel M, Kispal G, Lill R.

J Biol Chem. 2017 Dec 15;292(50):20656. doi: 10.1074/jbc.AAC117.000997. No abstract available.


Structure and functional dynamics of the mitochondrial Fe/S cluster synthesis complex.

Boniecki MT, Freibert SA, Mühlenhoff U, Lill R, Cygler M.

Nat Commun. 2017 Nov 3;8(1):1287. doi: 10.1038/s41467-017-01497-1.


Cryptococcus neoformans Iron-Sulfur Protein Biogenesis Machinery Is a Novel Layer of Protection against Cu Stress.

Garcia-Santamarina S, Uzarska MA, Festa RA, Lill R, Thiele DJ.

MBio. 2017 Oct 31;8(5). pii: e01742-17. doi: 10.1128/mBio.01742-17.


A novel de novo dominant mutation in ISCU associated with mitochondrial myopathy.

Legati A, Reyes A, Ceccatelli Berti C, Stehling O, Marchet S, Lamperti C, Ferrari A, Robinson AJ, Mühlenhoff U, Lill R, Zeviani M, Goffrini P, Ghezzi D.

J Med Genet. 2017 Dec;54(12):815-824. doi: 10.1136/jmedgenet-2017-104822. Epub 2017 Oct 27.


Isa1p is a component of the mitochondrial machinery for maturation of cellular iron-sulfur proteins and requires conserved cysteine residues for function.

Kaut A, Lange H, Diekert K, Kispal G, Lill R.

J Biol Chem. 2017 Oct 27;292(43):17980. doi: 10.1074/jbc.AAC117.000256. No abstract available.


Specialized function of yeast Isa1 and Isa2 proteins in the maturation of mitochondrial [4Fe-4S] proteins.

Mühlenhoff U, Richter N, Pines O, Pierik AJ, Lill R.

J Biol Chem. 2017 Oct 27;292(43):17979. doi: 10.1074/jbc.AAC117.000255. No abstract available.


Cellular requirements for iron-sulfur cluster insertion into the antiviral radical SAM protein viperin.

Upadhyay AS, Stehling O, Panayiotou C, Rösser R, Lill R, Överby AK.

J Biol Chem. 2017 Aug 18;292(33):13879-13889. doi: 10.1074/jbc.M117.780122. Epub 2017 Jun 14.


Iron-sulfur cluster biogenesis and trafficking in mitochondria.

Braymer JJ, Lill R.

J Biol Chem. 2017 Aug 4;292(31):12754-12763. doi: 10.1074/jbc.R117.787101. Epub 2017 Jun 14. Review.


The diferric-tyrosyl radical cluster of ribonucleotide reductase and cytosolic iron-sulfur clusters have distinct and similar biogenesis requirements.

Li H, Stümpfig M, Zhang C, An X, Stubbe J, Lill R, Huang M.

J Biol Chem. 2017 Jul 7;292(27):11445-11451. doi: 10.1074/jbc.M117.786178. Epub 2017 May 17.


Evolutionary conservation and in vitro reconstitution of microsporidian iron-sulfur cluster biosynthesis.

Freibert SA, Goldberg AV, Hacker C, Molik S, Dean P, Williams TA, Nakjang S, Long S, Sendra K, Bill E, Heinz E, Hirt RP, Lucocq JM, Embley TM, Lill R.

Nat Commun. 2017 Jan 4;8:13932. doi: 10.1038/ncomms13932.


Role of Nfu1 and Bol3 in iron-sulfur cluster transfer to mitochondrial clients.

Melber A, Na U, Vashisht A, Weiler BD, Lill R, Wohlschlegel JA, Winge DR.

Elife. 2016 Aug 17;5. pii: e15991. doi: 10.7554/eLife.15991.


Mitochondrial Bol1 and Bol3 function as assembly factors for specific iron-sulfur proteins.

Uzarska MA, Nasta V, Weiler BD, Spantgar F, Ciofi-Baffoni S, Saviello MR, Gonnelli L, Mühlenhoff U, Banci L, Lill R.

Elife. 2016 Aug 17;5. pii: e16673. doi: 10.7554/eLife.16673.


The conserved protein Dre2 uses essential [2Fe-2S] and [4Fe-4S] clusters for its function in cytosolic iron-sulfur protein assembly.

Netz DJ, Genau HM, Weiler BD, Bill E, Pierik AJ, Lill R.

Biochem J. 2016 Jul 15;473(14):2073-85. doi: 10.1042/BCJ20160416. Epub 2016 May 10.


Defects in Mitochondrial Iron-Sulfur Cluster Assembly Induce Cysteine S-Polythiolation on Iron-Sulfur Apoproteins.

Christ S, Leichert LI, Willms A, Lill R, Mühlenhoff U.

Antioxid Redox Signal. 2016 Jul 1;25(1):28-40. doi: 10.1089/ars.2015.6599. Epub 2016 Apr 22.


The mitochondrial monothiol glutaredoxin S15 is essential for iron-sulfur protein maturation in Arabidopsis thaliana.

Moseler A, Aller I, Wagner S, Nietzel T, Przybyla-Toscano J, Mühlenhoff U, Lill R, Berndt C, Rouhier N, Schwarzländer M, Meyer AJ.

Proc Natl Acad Sci U S A. 2015 Nov 3;112(44):13735-40. doi: 10.1073/pnas.1510835112. Epub 2015 Oct 19.


The deca-GX3 proteins Yae1-Lto1 function as adaptors recruiting the ABC protein Rli1 for iron-sulfur cluster insertion.

Paul VD, Mühlenhoff U, Stümpfig M, Seebacher J, Kugler KG, Renicke C, Taxis C, Gavin AC, Pierik AJ, Lill R.

Elife. 2015 Jul 16;4:e08231. doi: 10.7554/eLife.08231.


Mechanisms of intracellular compartmentalization and disease.

Lill R.

Eur J Cell Biol. 2015 Jul-Sep;94(7-9):277-9. doi: 10.1016/j.ejcb.2015.06.008. Epub 2015 Jun 19. No abstract available.


Compartmentalization of iron between mitochondria and the cytosol and its regulation.

Mühlenhoff U, Hoffmann B, Richter N, Rietzschel N, Spantgar F, Stehling O, Uzarska MA, Lill R.

Eur J Cell Biol. 2015 Jul-Sep;94(7-9):292-308. doi: 10.1016/j.ejcb.2015.05.003. Epub 2015 May 30. Review.


The role of mitochondria and the CIA machinery in the maturation of cytosolic and nuclear iron-sulfur proteins.

Lill R, Dutkiewicz R, Freibert SA, Heidenreich T, Mascarenhas J, Netz DJ, Paul VD, Pierik AJ, Richter N, Stümpfig M, Srinivasan V, Stehling O, Mühlenhoff U.

Eur J Cell Biol. 2015 Jul-Sep;94(7-9):280-91. doi: 10.1016/j.ejcb.2015.05.002. Epub 2015 May 31. Review.


Mutation of the iron-sulfur cluster assembly gene IBA57 causes fatal infantile leukodystrophy.

Debray FG, Stümpfig C, Vanlander AV, Dideberg V, Josse C, Caberg JH, Boemer F, Bours V, Stevens R, Seneca S, Smet J, Lill R, van Coster R.

J Inherit Metab Dis. 2015 Nov;38(6):1147-53. doi: 10.1007/s10545-015-9857-1. Epub 2015 May 14.


Special issue on iron-sulfur proteins: Structure, function, biogenesis and diseases.

Lill R, Broderick JB, Dean DR.

Biochim Biophys Acta. 2015 Jun;1853(6):1251-2. doi: 10.1016/j.bbamcr.2015.03.001. Epub 2015 Mar 5. No abstract available.


Distribution of selenoglucosinolates and their metabolites in Brassica treated with sodium selenate.

Matich AJ, McKenzie MJ, Lill RE, McGhie TK, Chen RK, Rowan DD.

J Agric Food Chem. 2015 Feb 25;63(7):1896-905. doi: 10.1021/jf505963c. Epub 2015 Feb 10.


Fe/S protein assembly gene IBA57 mutation causes hereditary spastic paraplegia.

Lossos A, Stümpfig C, Stevanin G, Gaussen M, Zimmerman BE, Mundwiller E, Asulin M, Chamma L, Sheffer R, Misk A, Dotan S, Gomori JM, Ponger P, Brice A, Lerer I, Meiner V, Lill R.

Neurology. 2015 Feb 17;84(7):659-67. doi: 10.1212/WNL.0000000000001270. Epub 2015 Jan 21.


Biogenesis of cytosolic and nuclear iron-sulfur proteins and their role in genome stability.

Paul VD, Lill R.

Biochim Biophys Acta. 2015 Jun;1853(6):1528-39. doi: 10.1016/j.bbamcr.2014.12.018. Epub 2015 Jan 10. Review.


Structure of the Kti11/Kti13 heterodimer and its double role in modifications of tRNA and eukaryotic elongation factor 2.

Glatt S, Zabel R, Vonkova I, Kumar A, Netz DJ, Pierik AJ, Rybin V, Lill R, Gavin AC, Balbach J, Breunig KD, Müller CW.

Structure. 2015 Jan 6;23(1):149-160. doi: 10.1016/j.str.2014.11.008. Epub 2014 Dec 24.


The role of mitochondria in cytosolic-nuclear iron–sulfur protein biogenesis and in cellular iron regulation.

Lill R, Srinivasan V, Mühlenhoff U.

Curr Opin Microbiol. 2014 Dec;22:111-9. doi: 10.1016/j.mib.2014.09.015. Review.


The basic leucine zipper stress response regulator Yap5 senses high-iron conditions by coordination of [2Fe-2S] clusters.

Rietzschel N, Pierik AJ, Bill E, Lill R, Mühlenhoff U.

Mol Cell Biol. 2015 Jan;35(2):370-8. doi: 10.1128/MCB.01033-14. Epub 2014 Nov 3.


Functional reconstitution of mitochondrial Fe/S cluster synthesis on Isu1 reveals the involvement of ferredoxin.

Webert H, Freibert SA, Gallo A, Heidenreich T, Linne U, Amlacher S, Hurt E, Mühlenhoff U, Banci L, Lill R.

Nat Commun. 2014 Oct 31;5:5013. doi: 10.1038/ncomms6013.


Consumption of selenium-enriched broccoli increases cytokine production in human peripheral blood mononuclear cells stimulated ex vivo, a preliminary human intervention study.

Bentley-Hewitt KL, Chen RK, Lill RE, Hedderley DI, Herath TD, Matich AJ, McKenzie MJ.

Mol Nutr Food Res. 2014 Dec;58(12):2350-7. doi: 10.1002/mnfr.201400438. Epub 2014 Oct 13.


SnapShot: eukaryotic Fe-S protein biogenesis.

Paul VD, Lill R.

Cell Metab. 2014 Aug 5;20(2):384-384.e1. doi: 10.1016/j.cmet.2014.07.010. No abstract available.


Cytosolic iron-sulphur protein assembly is functionally conserved and essential in procyclic and bloodstream Trypanosoma brucei.

Basu S, Netz DJ, Haindrich AC, Herlerth N, Lagny TJ, Pierik AJ, Lill R, Lukeš J.

Mol Microbiol. 2014 Sep;93(5):897-910. doi: 10.1111/mmi.12706. Epub 2014 Jul 23.


Only functional localization is faithful localization.

Lill R.

Microb Cell. 2014 Apr 7;1(4):115-117. doi: 10.15698/mic2014.04.141. No abstract available.


Crystal structures of nucleotide-free and glutathione-bound mitochondrial ABC transporter Atm1.

Srinivasan V, Pierik AJ, Lill R.

Science. 2014 Mar 7;343(6175):1137-40. doi: 10.1126/science.1246729.


Mitochondrial iron-sulfur protein biogenesis and human disease.

Stehling O, Wilbrecht C, Lill R.

Biochimie. 2014 May;100:61-77. doi: 10.1016/j.biochi.2014.01.010. Epub 2014 Jan 23. Review.


Maturation of cytosolic and nuclear iron-sulfur proteins.

Netz DJ, Mascarenhas J, Stehling O, Pierik AJ, Lill R.

Trends Cell Biol. 2014 May;24(5):303-12. doi: 10.1016/j.tcb.2013.11.005. Epub 2013 Dec 3. Review.


Viperin is an iron-sulfur protein that inhibits genome synthesis of tick-borne encephalitis virus via radical SAM domain activity.

Upadhyay AS, Vonderstein K, Pichlmair A, Stehling O, Bennett KL, Dobler G, Guo JT, Superti-Furga G, Lill R, Överby AK, Weber F.

Cell Microbiol. 2014 Jun;16(6):834-48. doi: 10.1111/cmi.12241. Epub 2013 Dec 3.


The functional interaction of mitochondrial Hsp70s with the escort protein Zim17 is critical for Fe/S biogenesis and substrate interaction at the inner membrane preprotein translocase.

Lewrenz I, Rietzschel N, Guiard B, Lill R, van der Laan M, Voos W.

J Biol Chem. 2013 Oct 25;288(43):30931-43. doi: 10.1074/jbc.M113.465997. Epub 2013 Sep 12.


The role of mitochondria in cellular iron-sulfur protein biogenesis: mechanisms, connected processes, and diseases.

Stehling O, Lill R.

Cold Spring Harb Perspect Biol. 2013 Aug 1;5(8):a011312. doi: 10.1101/cshperspect.a011312. Review.

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