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Dinucleotide Degradation by REXO2 Maintains Promoter Specificity in Mammalian Mitochondria.

Nicholls TJ, Spåhr H, Jiang S, Siira SJ, Koolmeister C, Sharma S, Kauppila JHK, Jiang M, Kaever V, Rackham O, Chabes A, Falkenberg M, Filipovska A, Larsson NG, Gustafsson CM.

Mol Cell. 2019 Sep 27. pii: S1097-2765(19)30695-1. doi: 10.1016/j.molcel.2019.09.010. [Epub ahead of print]


TEFM regulates both transcription elongation and RNA processing in mitochondria.

Jiang S, Koolmeister C, Misic J, Siira S, Kühl I, Silva Ramos E, Miranda M, Jiang M, Posse V, Lytovchenko O, Atanassov I, Schober FA, Wibom R, Hultenby K, Milenkovic D, Gustafsson CM, Filipovska A, Larsson NG.

EMBO Rep. 2019 Jun;20(6). pii: e48101. doi: 10.15252/embr.201948101. Epub 2019 Apr 29.


Copy-choice recombination during mitochondrial L-strand synthesis causes DNA deletions.

Persson Ö, Muthukumar Y, Basu S, Jenninger L, Uhler JP, Berglund AK, McFarland R, Taylor RW, Gustafsson CM, Larsson E, Falkenberg M.

Nat Commun. 2019 Feb 15;10(1):759. doi: 10.1038/s41467-019-08673-5.


RNase H1 directs origin-specific initiation of DNA replication in human mitochondria.

Posse V, Al-Behadili A, Uhler JP, Clausen AR, Reyes A, Zeviani M, Falkenberg M, Gustafsson CM.

PLoS Genet. 2019 Jan 3;15(1):e1007781. doi: 10.1371/journal.pgen.1007781. eCollection 2019 Jan.


Separating and Segregating the Human Mitochondrial Genome.

Nicholls TJ, Gustafsson CM.

Trends Biochem Sci. 2018 Nov;43(11):869-881. doi: 10.1016/j.tibs.2018.08.007. Epub 2018 Sep 14. Review.


Topoisomerase 3α Is Required for Decatenation and Segregation of Human mtDNA.

Nicholls TJ, Nadalutti CA, Motori E, Sommerville EW, Gorman GS, Basu S, Hoberg E, Turnbull DM, Chinnery PF, Larsson NG, Larsson E, Falkenberg M, Taylor RW, Griffith JD, Gustafsson CM.

Mol Cell. 2018 Jan 4;69(1):9-23.e6. doi: 10.1016/j.molcel.2017.11.033. Epub 2017 Dec 28.


Correction for Szilagyi et al., "Cyclin-Dependent Kinase 8 Regulates Mitotic Commitment in Fission Yeast".

Szilagyi Z, Banyai G, Davila Lopez M, McInerny CJ, Gustafsson CM.

Mol Cell Biol. 2017 Aug 11;37(17). pii: e00262-17. doi: 10.1128/MCB.00262-17. Print 2017 Sep 1. No abstract available.


Cyclin C influences the timing of mitosis in fission yeast.

Banyai G, Szilagyi Z, Baraznenok V, Khorosjutina O, Gustafsson CM.

Mol Biol Cell. 2017 Jul 1;28(13):1738-1744. doi: 10.1091/mbc.E16-11-0787. Epub 2017 May 17.


An Adaptable High-Throughput Technology Enabling the Identification of Specific Transcription Modulators.

Bergbrede T, Hoberg E, Larsson NG, Falkenberg M, Gustafsson CM.

SLAS Discov. 2017 Apr;22(4):378-386. doi: 10.1177/2472555217690326. Epub 2017 Jan 31.


Mutations in mitochondrial DNA causing tubulointerstitial kidney disease.

Connor TM, Hoer S, Mallett A, Gale DP, Gomez-Duran A, Posse V, Antrobus R, Moreno P, Sciacovelli M, Frezza C, Duff J, Sheerin NS, Sayer JA, Ashcroft M, Wiesener MS, Hudson G, Gustafsson CM, Chinnery PF, Maxwell PH.

PLoS Genet. 2017 Mar 7;13(3):e1006620. doi: 10.1371/journal.pgen.1006620. eCollection 2017 Mar.


Nucleotide pools dictate the identity and frequency of ribonucleotide incorporation in mitochondrial DNA.

Berglund AK, Navarrete C, Engqvist MK, Hoberg E, Szilagyi Z, Taylor RW, Gustafsson CM, Falkenberg M, Clausen AR.

PLoS Genet. 2017 Feb 16;13(2):e1006628. doi: 10.1371/journal.pgen.1006628. eCollection 2017 Feb.


Human Mitochondrial Transcription Factor B2 Is Required for Promoter Melting during Initiation of Transcription.

Posse V, Gustafsson CM.

J Biol Chem. 2017 Feb 17;292(7):2637-2645. doi: 10.1074/jbc.M116.751008. Epub 2016 Dec 27.


POLRMT regulates the switch between replication primer formation and gene expression of mammalian mtDNA.

Kühl I, Miranda M, Posse V, Milenkovic D, Mourier A, Siira SJ, Bonekamp NA, Neumann U, Filipovska A, Polosa PL, Gustafsson CM, Larsson NG.

Sci Adv. 2016 Aug 5;2(8):e1600963. doi: 10.1126/sciadv.1600963. eCollection 2016 Aug.


MGME1 processes flaps into ligatable nicks in concert with DNA polymerase γ during mtDNA replication.

Uhler JP, Thörn C, Nicholls TJ, Matic S, Milenkovic D, Gustafsson CM, Falkenberg M.

Nucleic Acids Res. 2016 Jul 8;44(12):5861-71. doi: 10.1093/nar/gkw468. Epub 2016 May 24.


Mitochondrial transcription termination factor 1 directs polar replication fork pausing.

Shi Y, Posse V, Zhu X, Hyvärinen AK, Jacobs HT, Falkenberg M, Gustafsson CM.

Nucleic Acids Res. 2016 Jul 8;44(12):5732-42. doi: 10.1093/nar/gkw302. Epub 2016 Apr 25.


Maintenance and Expression of Mammalian Mitochondrial DNA.

Gustafsson CM, Falkenberg M, Larsson NG.

Annu Rev Biochem. 2016 Jun 2;85:133-60. doi: 10.1146/annurev-biochem-060815-014402. Epub 2016 Mar 24. Review.


Loss of the Mediator subunit Med20 affects transcription of tRNA and other non-coding RNA genes in fission yeast.

Carlsten JO, Zhu X, López MD, Samuelsson T, Gustafsson CM.

Biochim Biophys Acta. 2016 Feb;1859(2):339-47. doi: 10.1016/j.bbagrm.2015.11.007. Epub 2015 Nov 19.


Regulation of DNA replication at the end of the mitochondrial D-loop involves the helicase TWINKLE and a conserved sequence element.

Jemt E, Persson Ö, Shi Y, Mehmedovic M, Uhler JP, Dávila López M, Freyer C, Gustafsson CM, Samuelsson T, Falkenberg M.

Nucleic Acids Res. 2015 Oct 30;43(19):9262-75. doi: 10.1093/nar/gkv804. Epub 2015 Aug 7.


MEG3 long noncoding RNA regulates the TGF-β pathway genes through formation of RNA-DNA triplex structures.

Mondal T, Subhash S, Vaid R, Enroth S, Uday S, Reinius B, Mitra S, Mohammed A, James AR, Hoberg E, Moustakas A, Gyllensten U, Jones SJ, Gustafsson CM, Sims AH, Westerlund F, Gorab E, Kanduri C.

Nat Commun. 2015 Jul 24;6:7743. doi: 10.1038/ncomms8743.


Mediator tail subunits can form amyloid-like aggregates in vivo and affect stress response in yeast.

Zhu X, Chen L, Carlsten JO, Liu Q, Yang J, Liu B, Gustafsson CM.

Nucleic Acids Res. 2015 Sep 3;43(15):7306-14. doi: 10.1093/nar/gkv629. Epub 2015 Jul 2.


Simultaneous DNA and RNA Mapping of Somatic Mitochondrial Mutations across Diverse Human Cancers.

Stewart JB, Alaei-Mahabadi B, Sabarinathan R, Samuelsson T, Gorodkin J, Gustafsson CM, Larsson E.

PLoS Genet. 2015 Jun 30;11(6):e1005333. doi: 10.1371/journal.pgen.1005333. eCollection 2015 Jun.


The exonuclease activity of DNA polymerase γ is required for ligation during mitochondrial DNA replication.

Macao B, Uhler JP, Siibak T, Zhu X, Shi Y, Sheng W, Olsson M, Stewart JB, Gustafsson CM, Falkenberg M.

Nat Commun. 2015 Jun 22;6:7303. doi: 10.1038/ncomms8303.


TEFM is a potent stimulator of mitochondrial transcription elongation in vitro.

Posse V, Shahzad S, Falkenberg M, Hällberg BM, Gustafsson CM.

Nucleic Acids Res. 2015 Mar 11;43(5):2615-24. doi: 10.1093/nar/gkv105. Epub 2015 Feb 17.


Whole exome sequencing reveals mutations in NARS2 and PARS2, encoding the mitochondrial asparaginyl-tRNA synthetase and prolyl-tRNA synthetase, in patients with Alpers syndrome.

Sofou K, Kollberg G, Holmström M, Dávila M, Darin N, Gustafsson CM, Holme E, Oldfors A, Tulinius M, Asin-Cayuela J.

Mol Genet Genomic Med. 2015 Jan;3(1):59-68. doi: 10.1002/mgg3.115. Epub 2014 Oct 23.


In vivo occupancy of mitochondrial single-stranded DNA binding protein supports the strand displacement mode of DNA replication.

Miralles Fusté J, Shi Y, Wanrooij S, Zhu X, Jemt E, Persson Ö, Sabouri N, Gustafsson CM, Falkenberg M.

PLoS Genet. 2014 Dec 4;10(12):e1004832. doi: 10.1371/journal.pgen.1004832. eCollection 2014 Dec.


Mediator can regulate mitotic entry and direct periodic transcription in fission yeast.

Banyai G, Lopez MD, Szilagyi Z, Gustafsson CM.

Mol Cell Biol. 2014 Nov;34(21):4008-18. doi: 10.1128/MCB.00819-14. Epub 2014 Aug 25.


Essential genetic interactors of SIR2 required for spatial sequestration and asymmetrical inheritance of protein aggregates.

Song J, Yang Q, Yang J, Larsson L, Hao X, Zhu X, Malmgren-Hill S, Cvijovic M, Fernandez-Rodriguez J, Grantham J, Gustafsson CM, Liu B, Nyström T.

PLoS Genet. 2014 Jul 31;10(7):e1004539. doi: 10.1371/journal.pgen.1004539. eCollection 2014 Jul.


In vitro-reconstituted nucleoids can block mitochondrial DNA replication and transcription.

Farge G, Mehmedovic M, Baclayon M, van den Wildenberg SM, Roos WH, Gustafsson CM, Wuite GJ, Falkenberg M.

Cell Rep. 2014 Jul 10;8(1):66-74. doi: 10.1016/j.celrep.2014.05.046. Epub 2014 Jun 26.


Hereditary myopathy with early respiratory failure is associated with misfolding of the titin fibronectin III 119 subdomain.

Hedberg C, Toledo AG, Gustafsson CM, Larson G, Oldfors A, Macao B.

Neuromuscul Disord. 2014 May;24(5):373-9. doi: 10.1016/j.nmd.2014.02.003. Epub 2014 Feb 15.


The amino terminal extension of mammalian mitochondrial RNA polymerase ensures promoter specific transcription initiation.

Posse V, Hoberg E, Dierckx A, Shahzad S, Koolmeister C, Larsson NG, Wilhelmsson LM, Hällberg BM, Gustafsson CM.

Nucleic Acids Res. 2014 Apr;42(6):3638-47. doi: 10.1093/nar/gkt1397. Epub 2014 Jan 20.


The UbL protein UBTD1 stably interacts with the UBE2D family of E2 ubiquitin conjugating enzymes.

Uhler JP, Spåhr H, Farge G, Clavel S, Larsson NG, Falkenberg M, Samuelsson T, Gustafsson CM.

Biochem Biophys Res Commun. 2014 Jan 3;443(1):7-12. doi: 10.1016/j.bbrc.2013.10.137. Epub 2013 Nov 6.


The multitalented Mediator complex.

Carlsten JO, Zhu X, Gustafsson CM.

Trends Biochem Sci. 2013 Nov;38(11):531-7. doi: 10.1016/j.tibs.2013.08.007. Epub 2013 Sep 25. Review.


Emerging roles of Cdk8 in cell cycle control.

Szilagyi Z, Gustafsson CM.

Biochim Biophys Acta. 2013 Sep;1829(9):916-20. doi: 10.1016/j.bbagrm.2013.04.010. Epub 2013 May 1. Review.


MTERF1 binds mtDNA to prevent transcriptional interference at the light-strand promoter but is dispensable for rRNA gene transcription regulation.

Terzioglu M, Ruzzenente B, Harmel J, Mourier A, Jemt E, López MD, Kukat C, Stewart JB, Wibom R, Meharg C, Habermann B, Falkenberg M, Gustafsson CM, Park CB, Larsson NG.

Cell Metab. 2013 Apr 2;17(4):618-26. doi: 10.1016/j.cmet.2013.03.006.


In vivo mutagenesis reveals that OriL is essential for mitochondrial DNA replication.

Wanrooij S, Miralles Fusté J, Stewart JB, Wanrooij PH, Samuelsson T, Larsson NG, Gustafsson CM, Falkenberg M.

EMBO Rep. 2012 Dec;13(12):1130-7. doi: 10.1038/embor.2012.161. Epub 2012 Oct 23.


Mammalian transcription factor A is a core component of the mitochondrial transcription machinery.

Shi Y, Dierckx A, Wanrooij PH, Wanrooij S, Larsson NG, Wilhelmsson LM, Falkenberg M, Gustafsson CM.

Proc Natl Acad Sci U S A. 2012 Oct 9;109(41):16510-5. doi: 10.1073/pnas.1119738109. Epub 2012 Sep 24.


A hybrid G-quadruplex structure formed between RNA and DNA explains the extraordinary stability of the mitochondrial R-loop.

Wanrooij PH, Uhler JP, Shi Y, Westerlund F, Falkenberg M, Gustafsson CM.

Nucleic Acids Res. 2012 Nov 1;40(20):10334-44. doi: 10.1093/nar/gks802. Epub 2012 Sep 10.


Structure of the human MTERF4-NSUN4 protein complex that regulates mitochondrial ribosome biogenesis.

Spåhr H, Habermann B, Gustafsson CM, Larsson NG, Hallberg BM.

Proc Natl Acad Sci U S A. 2012 Sep 18;109(38):15253-8. Epub 2012 Sep 4.


Protein sliding and DNA denaturation are essential for DNA organization by human mitochondrial transcription factor A.

Farge G, Laurens N, Broekmans OD, van den Wildenberg SM, Dekker LC, Gaspari M, Gustafsson CM, Peterman EJ, Falkenberg M, Wuite GJ.

Nat Commun. 2012;3:1013. doi: 10.1038/ncomms2001.


Mediator promotes CENP-a incorporation at fission yeast centromeres.

Carlsten JO, Szilagyi Z, Liu B, Lopez MD, Szászi E, Djupedal I, Nyström T, Ekwall K, Gustafsson CM, Zhu X.

Mol Cell Biol. 2012 Oct;32(19):4035-43. doi: 10.1128/MCB.00374-12. Epub 2012 Jul 30. Erratum in: Mol Cell Biol. 2012 Dec;32(24):5151.


Cyclin-dependent kinase 8 regulates mitotic commitment in fission yeast.

Szilagyi Z, Banyai G, Lopez MD, McInerny CJ, Gustafsson CM.

Mol Cell Biol. 2012 Jun;32(11):2099-109. doi: 10.1128/MCB.06316-11. Epub 2012 Mar 26. Erratum in: Mol Cell Biol. 2017 Aug 11;37(17 ):.


Role of human DNA glycosylase Nei-like 2 (NEIL2) and single strand break repair protein polynucleotide kinase 3'-phosphatase in maintenance of mitochondrial genome.

Mandal SM, Hegde ML, Chatterjee A, Hegde PM, Szczesny B, Banerjee D, Boldogh I, Gao R, Falkenberg M, Gustafsson CM, Sarkar PS, Hazra TK.

J Biol Chem. 2012 Jan 20;287(4):2819-29. doi: 10.1074/jbc.M111.272179. Epub 2011 Nov 30.


LRPPRC is necessary for polyadenylation and coordination of translation of mitochondrial mRNAs.

Ruzzenente B, Metodiev MD, Wredenberg A, Bratic A, Park CB, Cámara Y, Milenkovic D, Zickermann V, Wibom R, Hultenby K, Erdjument-Bromage H, Tempst P, Brandt U, Stewart JB, Gustafsson CM, Larsson NG.

EMBO J. 2012 Jan 18;31(2):443-56. doi: 10.1038/emboj.2011.392. Epub 2011 Nov 1.


Adenosine kinase deficiency disrupts the methionine cycle and causes hypermethioninemia, encephalopathy, and abnormal liver function.

Bjursell MK, Blom HJ, Cayuela JA, Engvall ML, Lesko N, Balasubramaniam S, Brandberg G, Halldin M, Falkenberg M, Jakobs C, Smith D, Struys E, von Döbeln U, Gustafsson CM, Lundeberg J, Wedell A.

Am J Hum Genet. 2011 Oct 7;89(4):507-15. doi: 10.1016/j.ajhg.2011.09.004. Epub 2011 Sep 28.


The mitochondrial DNA helicase TWINKLE can assemble on a closed circular template and support initiation of DNA synthesis.

Jemt E, Farge G, Bäckström S, Holmlund T, Gustafsson CM, Falkenberg M.

Nucleic Acids Res. 2011 Nov;39(21):9238-49. doi: 10.1093/nar/gkr653. Epub 2011 Aug 12.


Histone modifications influence mediator interactions with chromatin.

Zhu X, Zhang Y, Bjornsdottir G, Liu Z, Quan A, Costanzo M, Dávila López M, Westholm JO, Ronne H, Boone C, Gustafsson CM, Myers LC.

Nucleic Acids Res. 2011 Oct;39(19):8342-54. doi: 10.1093/nar/gkr551. Epub 2011 Jul 8.


MTERF4 regulates translation by targeting the methyltransferase NSUN4 to the mammalian mitochondrial ribosome.

Cámara Y, Asin-Cayuela J, Park CB, Metodiev MD, Shi Y, Ruzzenente B, Kukat C, Habermann B, Wibom R, Hultenby K, Franz T, Erdjument-Bromage H, Tempst P, Hallberg BM, Gustafsson CM, Larsson NG.

Cell Metab. 2011 May 4;13(5):527-39. doi: 10.1016/j.cmet.2011.04.002.


Mediator influences telomeric silencing and cellular life span.

Zhu X, Liu B, Carlsten JO, Beve J, Nyström T, Myers LC, Gustafsson CM.

Mol Cell Biol. 2011 Jun;31(12):2413-21. doi: 10.1128/MCB.05242-11. Epub 2011 Apr 11.


G-quadruplex structures in RNA stimulate mitochondrial transcription termination and primer formation.

Wanrooij PH, Uhler JP, Simonsson T, Falkenberg M, Gustafsson CM.

Proc Natl Acad Sci U S A. 2010 Sep 14;107(37):16072-7. doi: 10.1073/pnas.1006026107. Epub 2010 Aug 26.


LRPPRC is a mitochondrial matrix protein that is conserved in metazoans.

Sterky FH, Ruzzenente B, Gustafsson CM, Samuelsson T, Larsson NG.

Biochem Biophys Res Commun. 2010 Aug 6;398(4):759-64. doi: 10.1016/j.bbrc.2010.07.019. Epub 2010 Jul 13.


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