Skip to main content
H. Joseph Yost

H. Joseph Yost, PhD

Languages spoken: English

Academic Information

Departments Primary - Neurobiology , Adjunct - Pediatrics

Divisions: Hematology/Oncology

Academic Office Information

H. Joseph Yost, Ph.D. is Vice Chairman for Basic Science Research, Department of Pediatrics, Richard L. Stimson Presidential Endowed Chair, and Professor of Neurobiology & Anatomy

Dr. Yost works at the intersection between model organism genetics and the discovery of novel disease-causing mutations in human genomes. His long-term research goal is to understand the gene regulatory networks and developmental mechanisms that assign different cell identities in functionally appropriate positions in the vertebrate embryo, and to utilize this knowledge for the advancement of human medicine. This includes building bioinformatics toolsthat are disease agnostic and species agnostic, that we have applied to human genomes as well as model organism genomes. His lab is recognized as a founder and leader in the field of vertebrate left-right (LR) development, discovering genetic pathways and mechanisms that convert bilateral symmetry to left-right asymmetry, including essential functional and/or structural asymmetries in the heart, brain and digestive system. Disruptions of LR asymmetry result in a large percentage of complex congenital heart defects affecting approximately thirty-five thousand births per year in the US. His lab has generated zebrafish genetic models of human congenital heart disease (CHD), ciliopathies, Kabuki syndrome, Roberts syndrome, Li-Fraumeni syndrome and rare/orphan diseases in pediatrics.

Education History

Other Training University of Utah School of Business
University of California, Berkeley
Postdoctoral Fellow
Doctoral Training University of Chicago
Creighton University

Selected Publications

Journal Article

  1. OConnor TM, Guaman MC, Randell KA, Keenan HT, Snowden J, Mack JW, Camp EA, Perez O, Chang ML, Myers AL, Nigrovic LE, OToole J, Reed JL, Reese J, Rosenberg AR, Slater AC, Wootton SH, Ziniel SI, Yost HJ, Murray KO, Shekerdemian L, Chumpitazi CE (2023). Impact of the COVID-19 pandemic on pediatric faculty: a report from nine academic institutions. Pediatr Res, 95(3), 775-784. (Read full article)
  2. Eisa-Beygi S, Hu MM, Kumar SN, Jeffery BE, Collery RF, Vo NJ, Lamichanne BS, Yost HJ, Veldman MB, Link BA (2023). Mesenchymal Stromal Cells Facilitate Tip Cell Fusion Downstream of BMP-Mediated Venous Angiogenesis-Brief Report. Arterioscler Thromb Vasc Biol, 43(7), e231-e237. (Read full article)
  3. Casey MA, Hill JT, Hoshijima K, Bryan CD, Gribble SL, Brown JT, Chien CB, Yost HJ, Kwan KM (2021). Shutdown corner, a large deletion mutant isolated from a haploid mutagenesis screen in zebrafish. G3 (Bethesda), 12(3). (Read full article)
  4. Wesoowski S, Lemmon G, Hernandez EJ, Henrie A, Miller TA, Weyhrauch D, Puchalski MD, Bray BE, Shah RU, Deshmukh VG, Delaney R, Yost HJ, Eilbeck K, Tristani-Firouzi M, Yandell M (2022). An explainable artificial intelligence approach for predicting cardiovascular outcomes using electronic health records. PLOS Digit Health, 1(1). (Read full article)
  5. Yates JD, Russell RC, Barton NJ, Yost HJ, Hill JT (2021). A simple and rapid method for enzymatic synthesis of CRISPR-Cas9 sgRNA libraries. Nucleic Acids Res, 49(22), e131. (Read full article)
  6. Parvez S, Herdman C, Beerens M, Chakraborti K, Harmer ZP, Yeh JJ, MacRae CA, Yost HJ, Peterson RT (2021). MIC-Drop: A platform for large-scale in vivo CRISPR screens. Science, 373(6559), 1146-1151. (Read full article)
  7. Ward T, Tai W, Morton S, Impens F, Van Damme P, Van Haver D, Timmerman E, Venturini G, Zhang K, Jang MY, Willcox JAL, Haghighi A, Gelb BD, Chung WK, Goldmuntz E, Porter GA Jr, Lifton RP, Brueckner M, Yost HJ, Bruneau BG, Gorham J, Kim Y, Pereira A, Homsy J, Benson CC, DePalma SR, Varland S, Chen CS, Arnesen T, Gevaert K, Seidman C, Seidman JG (2021). Mechanisms of Congenital Heart Disease Caused by NAA15 Haploinsufficiency. Circ Res, 128(8), 1156-1169. (Read full article)
  8. Capasso TL, Li B, Volek HJ, Khalid W, Rochon ER, Anbalagan A, Herdman C, Yost HJ, Villanueva FS, Kim K, Roman BL (2019). BMP10-mediated ALK1 signaling is continuously required for vascular development and maintenance. Angiogenesis, 23(2), 203-220. (Read full article)
  9. Edwards JJ, Rouillard AD, Fernandez NF, Wang Z, Lachmann A, Shankaran SS, Bisgrove BW, Demarest B, Turan N, Srivastava D, Bernstein D, Deanfield J, Giardini A, Porter G, Kim R, Roberts AE, Newburger JW, Goldmuntz E, Brueckner M, Lifton RP, Seidman CE, Chung WK, Tristani-Firouzi M, Yost HJ, Maayan A, Gelb BD (2020). Systems Analysis Implicates WAVE2 Complex in the Pathogenesis of Developmental Left-Sided Obstructive Heart Defects. JACC Basic Transl Sci, 5(4), 376-386. (Read full article)
  10. Jurynec MJ, Bai X, Bisgrove BW, Jackson H, Nechiporuk A, Palu RAS, Grunwald HA, Su YC, Hoshijima K, Yost HJ, Zon LI, Grunwald DJ (2019). The Paf1 complex and P-TEFb have reciprocal and antagonist roles in maintaining multipotent neural crest progenitors. Development, 146(24). (Read full article)
  11. Watkins WS, Hernandez EJ, Wesolowski S, Bisgrove BW, Sunderland RT, Lin E, Lemmon G, Demarest BL, Miller TA, Bernstein D, Brueckner M, Chung WK, Gelb BD, Goldmuntz E, Newburger JW, Seidman CE, Shen Y, Yost HJ, Yandell M, Tristani-Firouzi M (2019). De novo and recessive forms of congenital heart disease have distinct genetic and phenotypic landscapes. Nat Commun, 10(1), 4722. (Read full article)
  12. Serrano MLA, Demarest BL, Tone-Pah-Hote T, Tristani-Firouzi M, Yost HJ (2019). Inhibition of Notch signaling rescues cardiovascular development in Kabuki Syndrome. PLoS Biol, 17(9), e3000087. (Read full article)
  13. Abdul-Wajid S, Demarest BL, Yost HJ (2018). Loss of embryonic neural crest derived cardiomyocytes causes adult onset hypertrophic cardiomyopathy in zebrafish. Nat Commun, 9(1), 4603. (Read full article)
  14. Karanth S, Adams JD, Serrano MLA, Quittner-Strom EB, Simcox J, Villanueva CJ, Ozcan L, Holland WL, Yost HJ, Vella A, Schlegel A (2018). A Hepatocyte FOXN3-α Cell Glucagon Axis Regulates Fasting Glucose. Cell Rep, 24(2), 312-319. (Read full article)
  15. Matsunami N, Shanmugam H, Baird L, Stevens J, Byrne JL, Barnhart DC, Rau C, Feldkamp ML, Yoder BA, Leppert MF, Yost HJ, Brunelli L (2018). Germline but not somatic de novo mutations are common in human congenital diaphragmatic hernia. Birth Defects Res, 110(7), 610-617. (Read full article)
  16. Bisgrove BW, Su YC, Yost HJ (2017). Maternal Gdf3 is an obligatory cofactor in Nodal signaling for embryonic axis formation in zebrafish. Elife, 6. (Read full article)
  17. Jin SC, Homsy J, Zaidi S, Lu Q, Morton S, DePalma SR, Zeng X, Qi H, Chang W, Sierant MC, Hung WC, Haider S, Zhang J, Knight J, Bjornson RD, Castaldi C, Tikhonoa IR, Bilguvar K, Mane SM, Sanders SJ, Mital S, Russell MW, Gaynor JW, Deanfield J, Giardini A, Porter GA Jr, Srivastava D, Lo CW, Shen Y, Watkins WS, Yandell M, Yost HJ, Tristani-Firouzi M, Newburger JW, Roberts AE, Kim R, Zhao H, Kaltman JR, Goldmuntz E, Chung WK, Seidman JG, Gelb BD, Seidman CE, Lifton RP, Brueckner M (2017). Contribution of rare inherited and de novo variants in 2,871 congenital heart disease probands. Nat Genet, 49(11), 1593-1601. (Read full article)
  18. Hill JT, Demarest B, Gorsi B, Smith M, Yost HJ (2017). Heart morphogenesis gene regulatory networks revealed by temporal expression analysis. Development, 144(19), 3487-3498. (Read full article)
  19. Shankaran SS, Dahlem TJ, Bisgrove BW, Yost HJ, Tristani-Firouzi M (2017). CRISPR/Cas9-Directed Gene Editing for the Generation of Loss-of-Function Mutants in High-Throughput Zebrafish F(0) Screens. Curr Protoc Mol Biol, 119, 31.9.1-31.9.22. (Read full article)
  20. Lyozin GT, Kosaka Y, Bhattacharje G, Yost HJ, Brunelli L (2017). Direct Isolation of Seamless Mutant Bacterial Artificial Chromosomes. Curr Protoc Mol Biol, 118, 8.6.1-8.6.29. (Read full article)
  21. Gittenberger-de Groot AC, Hoppenbrouwers T, Miquerol L, Kosaka Y, Poelmann RE, Wisse LJ, Yost HJ, Jongbloed MR, Deruiter MC, Brunelli L (2016). 14-3-3epsilon controls multiple developmental processes in the mouse heart. Dev Dyn, 245(11), 1107-1123. (Read full article)
  22. Merchant SS, Kosaka Y, Yost HJ, Hsu EW, Brunelli L (2016). Micro-Computed Tomography for the Quantitative 3-Dimensional Assessment of the Compact Myocardium in the Mouse Embryo. Circ J, 80(8), 1795-803. (Read full article)
  23. Karanth S, Zinkhan EK, Hill JT, Yost HJ, Schlegel A (2016). FOXN3 Regulates Hepatic Glucose Utilization. Cell Rep, 15(12), 2745-55. (Read full article)
  24. Bowles NE, Jou CJ, Arrington CB, Kennedy BJ, Earl A, Matsunami N, Meyers LL, Etheridge SP, Saarel EV, Bleyl SB, Yost HJ, Yandell M, Leppert MF, Tristani-Firouzi M, Gruber PJ, Baylor Hopkins Centers for Mendelian Genomics (2015). Exome analysis of a family with Wolff-Parkinson-White syndrome identifies a novel disease locus. Am J Med Genet A, 167A(12), 2975-84. (Read full article)
  25. Percival SM, Thomas HR, Amsterdam A, Carroll AJ, Lees JA, Yost HJ, Parant JM (2015). Variations in dysfunction of sister chromatid cohesion in esco2 mutant zebrafish reflect the phenotypic diversity of Roberts syndrome. Dis Model Mech, 8(8), 941-55. (Read full article)
  26. Nash D, Arrington CB, Kennedy BJ, Yandell M, Wu W, Zhang W, Ware S, Jorde LB, Gruber PJ, Yost HJ, Bowles NE, Bleyl SB (2015). Shared Segment Analysis and Next-Generation Sequencing Implicates the Retinoic Acid Signaling Pathway in Total Anomalous Pulmonary Venous Return (TAPVR). PLoS One, 10(6), e0131514. (Read full article)
  27. Hill JT, Demarest BL, Bisgrove BW, Su YC, Smith M, Yost HJ (2014). Poly peak parser: Method and software for identification of unknown indels using sanger sequencing of polymerase chain reaction products. Dev Dyn, 243(12), 1632-6. (Read full article)
  28. Lyozin GT, Bressloff PC, Kumar A, Kosaka Y, Demarest BL, Yost HJ, Kuehn MR, Brunelli L (2014). Isolation of rare recombinants without using selectable markers for one-step seamless BAC mutagenesis. Nat Methods, 11(9), 966-970. (Read full article)
  29. Neugebauer JM, Yost HJ (2013). FGF signaling is required for brain left-right asymmetry and brain midline formation. Dev Biol, 386(1), 123-34. (Read full article)
  30. Samson SC, Ferrer T, Jou CJ, Sachse FB, Shankaran SS, Shaw RM, Chi NC, Tristani-Firouzi M, Yost HJ (2013). 3-OST-7 regulates BMP-dependent cardiac contraction. PLoS Biol, 11(12), e1001727. (Read full article)
  31. Peterson AG, Wang X, Yost HJ (2013). Dvr1 transfers left-right asymmetric signals from Kupffer's vesicle to lateral plate mesoderm in zebrafish. Dev Biol, 382(1), 198-208. (Read full article)
  32. Arrington CB, Peterson AG, Yost HJ (2013). Sdc2 and Tbx16 regulate Fgf2-dependent epithelial cell morphogenesis in the ciliated organ of asymmetry. Development, 140(19), 4102-9. (Read full article)
  33. Neugebauer JM, Cadwallader AB, Amack JD, Bisgrove BW, Yost HJ (2013). Differential roles for 3-OSTs in the regulation of cilia length and motility. Development, 140(18), 3892-902. (Read full article)
  34. Hill JT, Demarest BL, Bisgrove BW, Gorsi B, Su YC, Yost HJ (2013). MMAPPR: mutation mapping analysis pipeline for pooled RNA-seq. Genome Res, 23(4), 687-97. (Read full article)
  35. Maguire CT, Demarest BL, Hill JT, Palmer JD, Brothman AR, Yost HJ, Condic ML (2013). Genome-wide analysis reveals the unique stem cell identity of human amniocytes. PLoS One, 8(1), e53372. (Read full article)
  36. Kosaka Y, Cieslik KA, Li L, Lezin G, Maguire CT, Saijoh Y, Toyo-oka K, Gambello MJ, Vatta M, Wynshaw-Boris A, Baldini A, Yost HJ, Brunelli L (2012). 14-3-3ε plays a role in cardiac ventricular compaction by regulating the cardiomyocyte cell cycle. Mol Cell Biol, 32(24), 5089-102. (Read full article)
  37. Arrington CB, Bleyl SB, Matsunami N, Bowles NE, Leppert TI, Demarest BL, Osborne K, Yoder BA, Byrne JL, Schiffman JD, Null DM, DiGeronimo R, Rollins M, Faix R, Comstock J, Camp NJ, Leppert MF, Yost HJ, Brunelli L (2012). A family-based paradigm to identify candidate chromosomal regions for isolated congenital diaphragmatic hernia. Am J Med Genet A, 158A(12), 3137-47. (Read full article)
  38. Cadwalader EL, Condic ML, Yost HJ (2012). 2-O-sulfotransferase regulates Wnt signaling, cell adhesion and cell cycle during zebrafish epiboly. Development, 139(7), 1296-305. (Read full article)
  39. Bisgrove BW, Makova S, Yost HJ, Brueckner M (2011). RFX2 is essential in the ciliated organ of asymmetry and an RFX2 transgene identifies a population of ciliated cells sufficient for fluid flow. Dev Biol, 363(1), 166-78. (Read full article)
  40. Wythe JD, Jurynec MJ, Urness LD, Jones CA, Sabeh MK, Werdich AA, Sato M, Yost HJ, Grunwald DJ, Macrae CA, Li DY (2011). Hadp1, a newly identified pleckstrin homology domain protein, is required for cardiac contractility in zebrafish. Dis Model Mech, 4(5), 607-21. (Read full article)
  41. Lezin G, Kosaka Y, Yost HJ, Kuehn MR, Brunelli L (2011). A one-step miniprep for the isolation of plasmid DNA and lambda phage particles. PLoS One, 6(8), e23457. (Read full article)
  42. Parant JM, George SA, Holden JA, Yost HJ (2010). Genetic modeling of Li-Fraumeni syndrome in zebrafish. Dis Model Mech, 3(1-2), 45-56. (Read full article)
  43. Parant JM, George SA, Pryor R, Wittwer CT, Yost HJ (2009). A rapid and efficient method of genotyping zebrafish mutants. Dev Dyn, 238(12), 3168-74. (Read full article)
  44. Arrington CB, Yost HJ (2009). Extra-embryonic syndecan 2 regulates organ primordia migration and fibrillogenesis throughout the zebrafish embryo. Development, 136(18), 3143-52. (Read full article)
  45. Neugebauer JM, Amack JD, Peterson AG, Bisgrove BW, Yost HJ (2009). FGF signalling during embryo development regulates cilia length in diverse epithelia. Nature, 458(7238), 651-4. (Read full article)
  46. Wang X, Yost HJ (2008). Initiation and propagation of posterior to anterior (PA) waves in zebrafish left-right development. Dev Dyn, 237(12), 3640-7. (Read full article)
  47. Kwan KM, Fujimoto E, Grabher C, Mangum BD, Hardy ME, Campbell DS, Parant JM, Yost HJ, Kanki JP, Chien CB (2007). The Tol2kit: a multisite gateway-based construction kit for Tol2 transposon transgenesis constructs. Dev Dyn, 236(11), 3088-99. (Read full article)
  48. Amack JD, Wang X, Yost HJ (2007). Two T-box genes play independent and cooperative roles to regulate morphogenesis of ciliated Kupffer's vesicle in zebrafish. Dev Biol, 310(2), 196-210. (Read full article)


  1. Poulain FE, Yost HJ (2015). Heparan sulfate proteoglycans: a sugar code for vertebrate development? [Review]. Development, 142(20), 3456-67. (Read full article)
  2. Kramer KL, Yost HJ (2003). Cardiac left-right development: are the early steps conserved? [Review]. Cold Spring Harb Symp Quant Biol, 67, 37-43. (Read full article)


  1. Sable C, Li JS, Tristani-Firouzi M, Fagerlin A, Silver RM, Yandel M, Yost HJ, Beaton A, Dale J, Engel M, Watkins D, Spurney C, Skinner AC, Armstrong SC, Shah SH, Allen N, Davis M, Hou L, Van Horn L, Labarthe D, Lloyd-Jones D, Marino B (2023). American Heart Association's Children's Strategically Focused Research Network Experience. J Am Heart Assoc, pp. e028356. (Read full article)