Publications

(Current lab members in bold.)

2024

• Wilson MH, Hensley MR, Shen MC, Lu HY, Quinlivan VH, Busch-Nentwich EM, Rawls JF, Farber SA (2024)

  Zebrafish are resilient to the loss of major diacylglycerol acyltransferase enzymes

  J Biol Chem
• Thierer JH, Foresti O, Yadav PK, Wilson MH, Moll TOC, Shen MC, Busch-Nentwich EM, Morash M, Mohlke KL, Rawls JF, Malhotra V, Hussain MM, Farber SA (2024)

  Pla2g12b drives expansion of triglyceride-rich lipoproteins

  Nat Commun
• Töpf A, Cox D, Zaharieva IT, Di Leo V, Sarparanta J, Jonson PH, Sealy IM, Smolnikov A, White RJ, Vihola A, Savarese M, Merteroglu M, Wali N, Laricchia KM, Venturini C, Vroling B, Stenton SL, Cummings BB, Harris E, Marini-Bettolo C, Diaz-Manera J, Henderson M, Barresi R, Duff J, England EM, Patrick J, Al-Husayni S, Biancalana V, Beggs AH, Bodi I, Bommireddipalli S, Bönnemann CG, Cairns A, Chiew M-T, Claeys KG, Cooper ST, Davis MR, Donkervoort S, Erasmus CE, Fassad MR, Genetti CA, Grosmann C, Jungbluth H, Kamsteeg E-J, Lornage X, Löscher WN, Malfatti E, Manzur A, Martí P, Mongini TE, Muelas N, Nishikawa A, O’Donnell-Luria A, Ogonuki N, O’Grady GL, O’Heir E, Paquay S, Phadke R, Pletcher BA, Romero NB, Schouten M, Shah S, Smuts I, Sznajer Y, Tasca G, Taylor RW, Tuite A, Van den Bergh P, VanNoy G, Voermans NC, Wanschitz JV, Wraige E, Yoshimura K, Oates EC, Nakagawa O, Nishino I, Laporte J, Vilchez JJ, MacArthur DG, Sarkozy A, Cordell HJ, Udd B, Busch-Nentwich EM, Muntoni F, Straub V (2024)

  Digenic inheritance involving a muscle-specific protein kinase and the giant titin protein causes a skeletal muscle myopathy

  Nat Genet
• Torraca V, White RJ, Sealy IM, Mazon-Moya M, Duggan G, Willis AR, Busch-Nentwich EM, Mostowy S (2024)

  Transcriptional profiling of zebrafish identifies host factors controlling susceptibility to Shigella flexneri

  Dis Model Mech

2023

• Schoenmakers E, Marelli F, Jørgensen HF, Visser WE, Moran C, Groeneweg S, Avalos C, Jurgens SJ, Figg N, Finigan A, Wali N, Agostini M, Wardle-Jones H, Lyons G, Rusk R, Gopalan D, Twiss P, Visser JJ, Goddard M, Nashef SAM, Heijmen R, Clift P, Sinha S, Pirruccello JP, Ellinor PT, Busch-Nentwich EM, Ramirez-Solis E, Murphy MP, Persani L, Bennett M, Chatterjee K (2023)

  Selenoprotein deficiency disorder predisposes to aortic aneurysm formation

  Nat Commun
• Fan J, Hale VL, Lelieveld LT, Whitworth LJ, Busch-Nentwich EM, Troll M, Edelstein PH, Cox TM, Roca JR, Aerts JMFG, Ramakrishnan L (2023)

  Gaucher disease protects against tuberculosis

  Proc Natl Acad Sci USA
• Banik I, Ghosh A, Beebe E, Burja B, Bertoncelj MF, Dooley CM, Markkanen E, Dummer R, Busch-Nentwich EM, Levesque MP (2023)

  P38 Mediates Tumor Suppression through Reduced Autophagy and Actin Cytoskeleton Changes in NRAS-Mutant Melanoma

  Cancers

2022

• Heppert JK, Lickwar CR, Tillman MC, Davis BR, Davison JM, Lu HY, Chen W, Busch-Nentwich EM, Corcoran DL, Rawls JF (2022)

  Conserved roles for Hnf4 family transcription factors in zebrafish development and intestinal function

  Genetics
• Pagán AJ, Lee LJ, Edwards-Hicks J, Moens CB, Tobin DM, Busch-Nentwich EM, Pearce EL, Ramakrishnan L (2022)

  mTOR-regulated mitochondrial metabolism limits mycobacterium-induced cytotoxicity

  Cell
• Tuschl K, White RJ, Trivedi C, Valdivia LE, Niklaus S, Bianco IH, Dadswell C, González-Méndez R, Sealy IM, Neuhauss SCF, Houart C, Rihel J, Wilson SW, Busch-Nentwich EM (2022)

  Loss of slc39a14 causes simultaneous manganese hypersensitivity and deficiency in zebrafish

  Dis Model Mech
• White RJ, Mackay E, Wilson SW, Busch-Nentwich EM (2022)

  Allele-specific gene expression can underlie altered transcript abundance in zebrafish mutants

  Elife
• Leggieri A, García-González J, Torres-Perez JV, Havelange W, Hosseinian S, Mech AM, Keatinge M, Busch-Nentwich EM, Brennan CH (2022)

  Ankk1 Loss of Function Disrupts Dopaminergic Pathways in Zebrafish

  Front Neurosci
• Sheardown E, Mech AM, Petrazzini MEM, Leggieri A, Gidziela A, Hosseinian S, Sealy IM, Torres-Perez JV, Busch-Nentwich EM, Malanchini M, Brennan CH (2022)

  Translational relevance of forward genetic screens in animal models for the study of psychiatric disease

  Neurosci Biobehav Rev
• Mech AM, Merteroglu M, Sealy IM, Teh MT, White RJ, Havelange W, Brennan CH, Busch-Nentwich EM (2022)

  Behavioral and Gene Regulatory Responses to Developmental Drug Exposures in Zebrafish

  Front Psychiatry
• Wali N, Merteroglu M, White RJ, Busch‐Nentwich EM (2022)

  Total Nucleic Acid Extraction from Single Zebrafish Embryos for Genotyping and RNA-seq

  Bio-protocol

2020

• Banik I, Cheng PF, Dooley CM, Travnickova J, Merteroglu M, Dummer R, Patton EE, Busch‐Nentwich EM, Levesque MP (2020)

  NRAS^Q61K melanoma tumor formation is reduced by p38‐MAPK14 activation in zebrafish models and NRAS‐mutated human melanoma cells

  Pigment Cell Melanoma Res
• Wilson MH, Rajan S, Danoff A, White RJ, Hensley MR, Quinlivan VH, Recacha R, Thierer JH, Tan FJ, Busch-Nentwich EM, Ruddock L, Hussain MM, Farber SA (2020)

  A point mutation decouples the lipid transfer activities of microsomal triglyceride transfer protein

  PLoS Genet
• Johansson JA, Marie KL, Lu Y, Brombin A, Santoriello C, Zeng Z, Zich J, Gautier P, von Kriegsheim A, Brunsdon H, Wheeler AP, Dreger M, Houston DR, Dooley CM, Sims AH, Busch-Nentwich EM, Zon LI, Illingworth RS, Patton EE (2020)

  PRL3-DDX21 Transcriptional Control of Endolysosomal Genes Restricts Melanocyte Stem Cell Differentiation

  Dev Cell
• García-González J, Brock AJ, Parker MO, Riley RJ, Jolliffe D, Sudwarts A, Teh MT, Busch-Nentwich EM, Stemple DL, Martineau AR, Kaprio J, Palviainen T, Kuan V, Walton RT, Brennan CH (2020)

  Identification of slit3 as a locus affecting nicotine preference in zebrafish and human smoking behaviour

  Elife

2019

• Coombs C, Georgantzoglou A, Walker HA, Patt J, Merten N, Poplimont H, Busch-Nentwich EM, Williams S, Kotsi C, Kostenis E, Sarris M (2019)

  Chemokine receptor trafficking coordinates neutrophil clustering and dispersal at wounds in zebrafish

  Nat Commun
• Collins JE, White RJ, Staudt N, Sealy IM, Packham I, Wali N, Tudor C, Mazzeo C, Green A, Siragher E, Ryder E, White JK, Papatheodoru I, Tang A, Fullgrabe A, Billis K, Geyer SH, Weninger WJ, Galli A, Hemberger M, Stemple DL, Robertson E, Smith JC, Mohun T, Adams DJ, Busch-Nentwich EM (2019)

  Common and distinct transcriptional signatures of mammalian embryonic lethality

  Nat Commun
• Dooley CM, Wali N, Sealy IM, White RJ, Stemple DL, Collins JE, Busch-Nentwich EM (2019)

  The gene regulatory basis of genetic compensation during neural crest induction

  PLoS Genet
• Weiner AMJ, Scampoli NL, Steeman TJ, Dooley CM, Busch-Nentwich EM, Kelsh RN, Calcaterra NB (2019)

  Dicer1 is required for pigment cell and craniofacial development in zebrafish

  Biochim Biophys Acta Gene Regul Mech
• Young RM, Hawkins TA, Cavodeassi F, Stickney HL, Schwarz Q, Lawrence LM, Wierzbicki C, Cheng BY, Luo J, Ambrosio EM, Klosner A, Sealy IM, Rowell J, Trivedi CA, Bianco IH, Allende ML, Busch-Nentwich EM, Gestri G, Wilson SW (2019)

  Compensatory growth renders Tcf7l1a dispensable for eye formation despite its requirement in eye field specification

  Elife
• Lush ME, Diaz DC, Koenecke N, Baek S, Boldt H, St Peter MK, Gaitan-Escudero T, Romero-Carvajal A, Busch-Nentwich EM, Perera AG, Hall KE, Peak A, Haug JS, Piotrowski T (2019)

  scRNA-Seq reveals distinct stem cell populations that drive hair cell regeneration after loss of Fgf and Notch signaling

  Elife

2018

• Minchin JEN, Scahill CM, Staudt N, Busch-Nentwich EM, Rawls JF (2018)

  Deep phenotyping in zebrafish reveals genetic and diet-induced adiposity changes that may inform disease risk

  J Lipid Res
• Perez-Garcia V, Fineberg E, Wilson R, Murray A, Mazzeo CI, Tudor C, Sienerth A, White JK, Tuck E, Ryder EJ, Gleeson D, Siragher E, Wardle-Jones H, Staudt N, Wali N, Collins J, Geyer S, Busch-Nentwich EM, Galli A, Smith JC, Robertson E, Adams DJ, Weninger WJ, Mohun T, Hemberger M (2018)

  Placentation defects are highly prevalent in embryonic lethal mouse mutants

  Nature

2017

• Scahill CM, Digby Z, Sealy IM, White RJ, Wali N, Collins JE, Stemple DL, Busch-Nentwich EM (2017)

  The age of heterozygous telomerase mutant parents influences the adult phenotype of their offspring irrespective of genotype in zebrafish

  Wellcome Open Res
• Anderson JL, Mulligan TS, Shen MC, Wang H, Scahill CM, Tan FJ, Du SJ, Busch-Nentwich EM, Farber SA. (2017)

  mRNA processing in mutant zebrafish lines generated by chemical and CRISPR-mediated mutagenesis produces unexpected transcripts that escape nonsense-mediated decay

  PLoS Genet
• White RJ, Collins JE, Sealy IM, Wali N, Dooley CM, Digby Z, Stemple DL, Murphy DN, Billis K, Hourlier T, Füllgrabe A, Davis MP, Enright AJ, Busch-Nentwich EM (2017)

  A high-resolution mRNA expression time course of embryonic development in zebrafish

  Elife
• Henke K, Daane JM, Hawkins MB, Dooley CM, Busch-Nentwich EM, Stemple DL, Harris MP (2017)

  Genetic Screen for Postembryonic Development in the Zebrafish (Danio rerio): Dominant Mutations Affecting Adult Form

  Genetics
• Scahill CM, Digby Z, Sealy IM, Wojciechowska S, White RJ, Collins JE, Stemple DL, Bartke T, Mathers ME, Patton EE, Busch-Nentwich EM (2017)

  Loss of the chromatin modifier Kdm2aa causes BrafV600E-independent spontaneous melanoma in zebrafish

  PLoS Genet
• Erickson T, Morgan CP, Olt J, Hardy K, Busch-Nentwich E, Maeda R, Clemens R, Krey JF, Nechiporuk A, Barr-Gillespie PG, Marcotti W, Nicolson T (2017)

  Integration of Tmc1/2 into the mechanotransduction complex in zebrafish hair cells is regulated by Transmembrane O-methyltransferase (Tomt)

  Elife
• Borgel J, Tyl M, Schiller K, Pusztai Z, Dooley CM, Deng W, Wooding C, White RJ, Warnecke T, Leonhardt H, Busch-Nentwich EM, Bartke T (2017)

  KDM2A integrates DNA and histone modification signals through a CXXC/PHD module and direct interaction with HP1

  Nucleic Acids Res

Selected Earlier Publications

• Brocal I, White RJ, Dooley CM, Carruthers SN, Clark R, Hall A, Busch-Nentwich EM, Stemple DL, Kettleborough RN (2016)

  Efficient identification of CRISPR/Cas9-induced insertions/deletions by direct germline screening in zebrafish

  BMC Genomics
• Chow W, Brugger K, Caccamo M, Sealy I, Torrance J, Howe K (2016)

  gEVAL - a web-based browser for evaluating genome assemblies

  Bioinformatics
• Collins JE, Wali N, Sealy IM, Morris JA, White RJ, Leonard SR, Jackson DK, Jones MC, Smerdon NC, Zamora J, Dooley CM, Carruthers SN, Barrett JC, Stemple DL, Busch-Nentwich EM (2015)

  High-throughput and quantitative genome-wide messenger RNA sequencing for molecular phenotyping

  BMC Genomics
• Dooley CM, Scahill C, Fényes F, Kettleborough RN, Stemple DL, Busch-Nentwich EM (2013)

  Multi-allelic phenotyping--a systematic approach for the simultaneous analysis of multiple induced mutations

  Methods
• Kettleborough RN, Busch-Nentwich EM, Harvey SA, Dooley CM, de Bruijn E, van Eeden F, Sealy I, White RJ, Herd C, Nijman IJ, Fényes F, Mehroke S, Scahill C, Gibbons R, Wali N, Carruthers S, Hall A, Yen J, Cuppen E, Stemple DL (2013)

  A systematic genome-wide analysis of zebrafish protein-coding gene function

  Nature
• Howe K, Clark MD, Torroja CF, Torrance J, Berthelot C, Muffato M, Collins JE, Humphray S, McLaren K, Matthews L, McLaren S, Sealy I, Caccamo M, Churcher C, Scott C, Barrett JC, Koch R, Rauch GJ, White S, Chow W, Kilian B, Quintais LT, Guerra-Assunção JA, Zhou Y, Gu Y, Yen J, Vogel JH, Eyre T, Redmond S, Banerjee R, Chi J, Fu B, Langley E, Maguire SF, Laird GK, Lloyd D, Kenyon E, Donaldson S, Sehra H, Almeida-King J, Loveland J, Trevanion S, Jones M, Quail M, Willey D, Hunt A, Burton J, Sims S, McLay K, Plumb B, Davis J, Clee C, Oliver K, Clark R, Riddle C, Elliot D, Threadgold G, Harden G, Ware D, Begum S, Mortimore B, Kerry G, Heath P, Phillimore B, Tracey A, Corby N, Dunn M, Johnson C, Wood J, Clark S, Pelan S, Griffiths G, Smith M, Glithero R, Howden P, Barker N, Lloyd C, Stevens C, Harley J, Holt K, Panagiotidis G, Lovell J, Beasley H, Henderson C, Gordon D, Auger K, Wright D, Collins J, Raisen C, Dyer L, Leung K, Robertson L, Ambridge K, Leongamornlert D, McGuire S, Gilderthorp R, Griffiths C, Manthravadi D, Nichol S, Barker G, Whitehead S, Kay M, Brown J, Murnane C, Gray E, Humphries M, Sycamore N, Barker D, Saunders D, Wallis J, Babbage A, Hammond S, Mashreghi-Mohammadi M, Barr L, Martin S, Wray P, Ellington A, Matthews N, Ellwood M, Woodmansey R, Clark G, Cooper J, Tromans A, Grafham D, Skuce C, Pandian R, Andrews R, Harrison E, Kimberley A, Garnett J, Fosker N, Hall R, Garner P, Kelly D, Bird C, Palmer S, Gehring I, Berger A, Dooley CM, Ersan-Ürün Z, Eser C, Geiger H, Geisler M, Karotki L, Kirn A, Konantz J, Konantz M, Oberländer M, Rudolph-Geiger S, Teucke M, Lanz C, Raddatz G, Osoegawa K, Zhu B, Rapp A, Widaa S, Langford C, Yang F, Schuster SC, Carter NP, Harrow J, Ning Z, Herrero J, Searle SM, Enright A, Geisler R, Plasterk RH, Lee C, Westerfield M, de Jong PJ, Zon LI, Postlethwait JH, Nüsslein-Volhard C, Hubbard TJ, Roest Crollius H, Rogers J, Stemple DL (2013)

  The zebrafish reference genome sequence and its relationship to the human genome

  Nature
• Jacoby AS, Busch-Nentwich E, Bryson-Richardson RJ, Hall TE, Berger J, Berger S, Sonntag C, Sachs C, Geisler R, Stemple DL, Currie PD (2009)

  The zebrafish dystrophic mutant softy maintains muscle fibre viability despite basement membrane rupture and muscle detachment

  Development
• Flicek P, Aken BL, Beal K, Ballester B, Caccamo M, Chen Y, Clarke L, Coates G, Cunningham F, Cutts T, Down T, Dyer SC, Eyre T, Fitzgerald S, Fernandez-Banet J, Gräf S, Haider S, Hammond M, Holland R, Howe KL, Howe K, Johnson N, Jenkinson A, Kähäri A, Keefe D, Kokocinski F, Kulesha E, Lawson D, Longden I, Megy K, Meidl P, Overduin B, Parker A, Pritchard B, Prlic A, Rice S, Rios D, Schuster M, Sealy I, Slater G, Smedley D, Spudich G, Trevanion S, Vilella AJ, Vogel J, White S, Wood M, Birney E, Cox T, Curwen V, Durbin R, Fernandez-Suarez XM, Herrero J, Hubbard TJ, Kasprzyk A, Proctor G, Smith J, Ureta-Vidal A, Searle S (2008)

  Ensembl 2008

  Nucleic Acids Res
• Geisler R, Rauch GJ, Geiger-Rudolph S, Albrecht A, van Bebber F, Berger A, Busch-Nentwich E, Dahm R, Dekens MP, Dooley C, Elli AF, Gehring I, Geiger H, Geisler M, Glaser S, Holley S, Huber M, Kerr A, Kirn A, Knirsch M, Konantz M, Küchler AM, Maderspacher F, Neuhauss SC, Nicolson T, Ober EA, Praeg E, Ray R, Rentzsch B, Rick JM, Rief E, Schauerte HE, Schepp CP, Schönberger U, Schonthaler HB, Seiler C, Sidi S, Söllner C, Wehner A, Weiler C, Nüsslein-Volhard C (2007)

  Large-scale mapping of mutations affecting zebrafish development

  BMC Genomics