Liao Lab
Current Preprints and Manuscripts
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Carroll SH, Schafer S, Kawasaki K, Tsimbal C, Jule A, Hallett S, Li E, Liao EC. dact1/2 modifies noncanonical Wnt signaling and calpain 8 expression to regulate convergent extension and craniofacial development. Preprint. bioRxiv. 2023;2023.11.07.566024. Published 2023 Nov 7. doi:10.1101/2023.11.07.566024
Peer Reviewed Articles
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Caetano da Silva C, Macias Trevino C, Mitchell J, Murali H, Tsimbal C, Dalessandro E, Carroll SH, Kochhar S, Curtis SW, Cheng CHE, Wang F, Kutschera E, Carstens RP, Xing Y, Wang K, Leslie EJ, Liao EC. Functional analysis of ESRP1/2 gene variants and CTNND1 isoforms in orofacial cleft pathogenesis. Commun Biol. 2024;7(1):1040. doi: 10.1038/s42003-024-06715-3. PMID: 39179789
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Murali H, Wang P, Liao EC, Wang K. Genetic variant classification by predicted protein structure: A case study on IRF6. Comput Struct Biotechnol J. 2024 Dec;23:892-904. doi: 10.1016/j.csbj.2024.01.019. eCollection 2024 Dec. PubMed PMID: 38370976; PubMed Central PMCID: PMC10869248.
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Yoon B, Yeung P, Santistevan N, Bluhm LE, Kawasaki K, Kueper J, Dubielzig R, VanOudenhove J, Cotney J, Liao EC, Grinblat Y. Zebrafish models of alx-linked frontonasal dysplasia reveal a role for Alx1 and Alx3 in the anterior segment and vasculature of the developing eye. Biol Open. 2022 May 15;11(5). doi: 10.1242/bio.059189. Epub 2022 Jun 7. PubMed PMID: 35142342; PubMed Central PMCID: PMC9167625.
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Kumari P, Sturgeon M, Bonde G, Cornell RA. Generating Zebrafish RNA-Less Mutant Alleles by Deleting Gene Promoters with CRISPR/Cas9. Methods Mol Biol. 2022;2403:91-106. doi: 10.1007/978-1-0716-1847-9_8. PubMed PMID: 34913119; PubMed Central PMCID: PMC10136374.
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Alhazmi N, Carroll SH, Kawasaki K, Woronowicz KC, Hallett SA, Macias Trevino C, Li EB, Baron R, Gori F, Yelick PC, Harris MP, Liao EC. Synergistic roles of Wnt modulators R-spondin2 and R-spondin3 in craniofacial morphogenesis and dental development. Sci Rep. 2021 Mar 12;11(1):5871. doi: 10.1038/s41598-021-85415-y. PubMed PMID: 33712657; PubMed Central PMCID: PMC7954795.
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Carroll SH, Macias Trevino C, Li EB, Kawasaki K, Myers N, Hallett SA, Alhazmi N, Cotney J, Carstens RP, Liao EC. An Irf6-Esrp1/2 regulatory axis controls midface morphogenesis in vertebrates. Development. 2020 Dec 23;147(24). doi: 10.1242/dev.194498. PubMed PMID: 33234718; PubMed Central PMCID: PMC7774891.
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Pini J, Kueper J, Hu YD, Kawasaki K, Yeung P, Tsimbal C, Yoon B, Carmichael N, Maas RL, Cotney J, Grinblat Y, Liao EC. ALX1-related frontonasal dysplasia results from defective neural crest cell development and migration. EMBO Mol Med. 2020 Oct 7;12(10):e12013. doi: 10.15252/emmm.202012013. Epub 2020 Sep 11. PubMed PMID: 32914578; PubMed Central PMCID: PMC7539331.
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Samuels BD, Aho R, Brinkley JF, Bugacov A, Feingold E, Fisher S, Gonzalez-Reiche AS, Hacia JG, Hallgrimsson B, Hansen K, Harris MP, Ho TV, Holmes G, Hooper JE, Jabs EW, Jones KL, Kesselman C, Klein OD, Leslie EJ, Li H, Liao EC, Long H, Lu N, Maas RL, Marazita ML, Mohammed J, Prescott S, Schuler R, Selleri L, Spritz RA, Swigut T, van Bakel H, Visel A, Welsh I, Williams C, Williams TJ, Wysocka J, Yuan Y, Chai Y. FaceBase 3: analytical tools and FAIR resources for craniofacial and dental research. Development. 2020 Sep 21;147(18). doi: 10.1242/dev.191213. Review. PubMed PMID: 32958507; PubMed Central PMCID: PMC7522026.
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Yang X, Jounaidi Y, Mukherjee K, Fantasia RJ, Liao EC, Yu B, Forman SA. Drug-selective Anesthetic Insensitivity of Zebrafish Lacking γ-Aminobutyric Acid Type A Receptor β3 Subunits. Anesthesiology. 2019 Dec;131(6):1276-1291. doi: 10.1097/ALN.0000000000002963. PubMed PMID: 31567362; PubMed Central PMCID: PMC6856434.
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Kousa YA, Zhu H, Fakhouri WD, Lei Y, Kinoshita A, Roushangar RR, Patel NK, Agopian AJ, Yang W, Leslie EJ, Busch TD, Mansour TA, Li X, Smith AL, Li EB, Sharma DB, Williams TJ, Chai Y, Amendt BA, Liao EC, Mitchell LE, Bassuk AG, Gregory S, Ashley-Koch A, Shaw GM, Finnell RH, Schutte BC. The TFAP2A-IRF6-GRHL3 genetic pathway is conserved in neurulation. Hum Mol Genet. 2019 May 15;28(10):1726-1737. doi: 10.1093/hmg/ddz010. PubMed PMID: 30689861; PubMed Central PMCID: PMC6494790.
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Mukherjee K, Liao EC. Generation and characterization of a zebrafish muscle specific inducible Cre line. Transgenic Res. 2018 Dec;27(6):559-569. doi: 10.1007/s11248-018-0098-6. Epub 2018 Oct 23. PubMed PMID: 30353407; PubMed Central PMCID: PMC6364321.
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Haghighi A, Krier JB, Toth-Petroczy A, Cassa CA, Frank NY, Carmichael N, Fieg E, Bjonnes A, Mohanty A, Briere LC, Lincoln S, Lucia S, Gupta VA, Söylemez O, Sutti S, Kooshesh K, Qiu H, Fay CJ, Perroni V, Valerius J, Hanna M, Frank A, Ouahed J, Snapper SB, Pantazi A, Chopra SS, Leshchiner I, Stitziel NO, Feldweg A, Mannstadt M, Loscalzo J, Sweetser DA, Liao E, Stoler JM, Nowak CB, Sanchez-Lara PA, Klein OD, Perry H, Patsopoulos NA, Raychaudhuri S, Goessling W, Green RC, Seidman CE, MacRae CA, Sunyaev SR, Maas RL, Vuzman D. An integrated clinical program and crowdsourcing strategy for genomic sequencing and Mendelian disease gene discovery. NPJ Genom Med. 2018;3:21. doi: 10.1038/s41525-018-0060-9. eCollection 2018. Review. PubMed PMID: 30131872; PubMed Central PMCID: PMC6089983
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Ishii K, Mukherjee K, Okada T, Liao EC. Genetic Requirement of talin1 for Proliferation of Cranial Neural Crest Cells during Palate Development. Plast Reconstr Surg Glob Open. 2018 Mar;6(3):e1633. doi: 10.1097/GOX.0000000000001633. eCollection 2018 Mar. PubMed PMID: 29707441; PubMed Central PMCID: PMC5908504.
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Duncan KM, Mukherjee K, Cornell RA, Liao EC. Zebrafish models of orofacial clefts. Dev Dyn. 2017 Nov;246(11):897-914. doi: 10.1002/dvdy.24566. Epub 2017 Sep 25. Review. PubMed PMID: 28795449; PubMed Central PMCID: PMC5777297
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Li EB, Truong D, Hallett SA, Mukherjee K, Schutte BC, Liao EC. Rapid functional analysis of computationally complex rare human IRF6 gene variants using a novel zebrafish model. PLoS Genet. 2017 Sep;13(9):e1007009. doi: 10.1371/journal.pgen.1007009. eCollection 2017 Sep. PubMed PMID: 28945736; PubMed Central PMCID: PMC5628943.
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Ciarlo C, Kaufman CK, Kinikoglu B, Michael J, Yang S, D Amato C, Blokzijl-Franke S, den Hertog J, Schlaeger TM, Zhou Y, Liao E, Zon LI. A chemical screen in zebrafish embryonic cells establishes that Akt activation is required for neural crest development. Elife. 2017 Aug 23;6. doi: 10.7554/eLife.29145. PubMed PMID: 28832322; PubMed Central PMCID: PMC5599238.
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Shaw ND, Brand H, Kupchinsky ZA, Bengani H, Plummer L, Jones TI, Erdin S, Williamson KA, Rainger J, Stortchevoi A, Samocha K, Currall BB, Dunican DS, Collins RL, Willer JR, Lek A, Lek M, Nassan M, Pereira S, Kammin T, Lucente D, Silva A, Seabra CM, Chiang C, An Y, Ansari M, Rainger JK, Joss S, Smith JC, Lippincott MF, Singh SS, Patel N, Jing JW, Law JR, Ferraro N, Verloes A, Rauch A, Steindl K, Zweier M, Scheer I, Sato D, Okamoto N, Jacobsen C, Tryggestad J, Chernausek S, Schimmenti LA, Brasseur B, Cesaretti C, García-Ortiz JE, Buitrago TP, Silva OP, Hoffman JD, Mühlbauer W, Ruprecht KW, Loeys BL, Shino M, Kaindl AM, Cho CH, Morton CC, Meehan RR, van Heyningen V, Liao EC, Balasubramanian R, Hall JE, Seminara SB, Macarthur D, Moore SA, Yoshiura KI, Gusella JF, Marsh JA, Graham JM Jr, Lin AE, Katsanis N, Jones PL, Crowley WF Jr, Davis EE, FitzPatrick DR, Talkowski ME. SMCHD1 mutations associated with a rare muscular dystrophy can also cause isolated arhinia and Bosma arhinia microphthalmia syndrome. Nat Genet. 2017 Feb;49(2):238-248. doi: 10.1038/ng.3743. Epub 2017 Jan 9. PubMed PMID: 28067909; PubMed Central PMCID: PMC5473428.
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Ling IT, Rochard L, Liao EC. Distinct requirements of wls, wnt9a, wnt5b and gpc4 in regulating chondrocyte maturation and timing of endochondral ossification. Dev Biol. 2017 Jan 15;421(2):219-232. doi: 10.1016/j.ydbio.2016.11.016. Epub 2016 Nov 29. PubMed PMID: 27908786; PubMed Central PMCID: PMC5266562.
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Redin C, Brand H, Collins RL, Kammin T, Mitchell E, Hodge JC, Hanscom C, Pillalamarri V, Seabra CM, Abbott MA, Abdul-Rahman OA, Aberg E, Adley R, Alcaraz-Estrada SL, Alkuraya FS, An Y, Anderson MA, Antolik C, Anyane-Yeboa K, Atkin JF, Bartell T, Bernstein JA, Beyer E, Blumenthal I, Bongers EM, Brilstra EH, Brown CW, Brüggenwirth HT, Callewaert B, Chiang C, Corning K, Cox H, Cuppen E, Currall BB, Cushing T, David D, Deardorff MA, Dheedene A, D'Hooghe M, de Vries BB, Earl DL, Ferguson HL, Fisher H, FitzPatrick DR, Gerrol P, Giachino D, Glessner JT, Gliem T, Grady M, Graham BH, Griffis C, Gripp KW, Gropman AL, Hanson-Kahn A, Harris DJ, Hayden MA, Hill R, Hochstenbach R, Hoffman JD, Hopkin RJ, Hubshman MW, Innes AM, Irons M, Irving M, Jacobsen JC, Janssens S, Jewett T, Johnson JP, Jongmans MC, Kahler SG, Koolen DA, Korzelius J, Kroisel PM, Lacassie Y, Lawless W, Lemyre E, Leppig K, Levin AV, Li H, Li H, Liao EC, Lim C, Lose EJ, Lucente D, Macera MJ, Manavalan P, Mandrile G, Marcelis CL, Margolin L, Mason T, Masser-Frye D, McClellan MW, Mendoza CJ, Menten B, Middelkamp S, Mikami LR, Moe E, Mohammed S, Mononen T, Mortenson ME, Moya G, Nieuwint AW, Ordulu Z, Parkash S, Pauker SP, Pereira S, Perrin D, Phelan K, Aguilar RE, Poddighe PJ, Pregno G, Raskin S, Reis L, Rhead W, Rita D, Renkens I, Roelens F, Ruliera J, Rump P, Schilit SL, Shaheen R, Sparkes R, Spiegel E, Stevens B, Stone MR, Tagoe J, Thakuria JV, van Bon BW, van de Kamp J, van Der Burgt I, van Essen T, van Ravenswaaij-Arts CM, van Roosmalen MJ, Vergult S, Volker-Touw CM, Warburton DP, Waterman MJ, Wiley S, Wilson A, Yerena-de Vega MC, Zori RT, Levy B, Brunner HG, de Leeuw N, Kloosterman WP, Thorland EC, Morton CC, Gusella JF, Talkowski ME. The genomic landscape of balanced cytogenetic abnormalities associated with human congenital anomalies. Nat Genet. 2017 Jan;49(1):36-45. doi: 10.1038/ng.3720. Epub 2016 Nov 14. PubMed PMID: 27841880; PubMed Central PMCID: PMC5307971
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Rochard L, Monica SD, Ling IT, Kong Y, Roberson S, Harland R, Halpern M, Liao EC. Roles of Wnt pathway genes wls, wnt9a, wnt5b, frzb and gpc4 in regulating convergent-extension during zebrafish palate morphogenesis. Development. 2016 Jul 15;143(14):2541-7. doi: 10.1242/dev.137000. Epub 2016 Jun 10. PubMed PMID: 27287801; PubMed Central PMCID: PMC4958341.
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Mukherjee K, Ishii K, Pillalamarri V, Kammin T, Atkin JF, Hickey SE, Xi QJ, Zepeda CJ, Gusella JF, Talkowski ME, Morton CC, Maas RL, Liao EC. Actin capping protein CAPZB regulates cell morphology, differentiation, and neural crest migration in craniofacial morphogenesis†. Hum Mol Genet. 2016 Apr 1;25(7):1255-70. doi: 10.1093/hmg/ddw006. Epub 2016 Jan 11. PubMed PMID: 26758871; PubMed Central PMCID: PMC4787901.
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Kaufman CK, Mosimann C, Fan ZP, Yang S, Thomas AJ, Ablain J, Tan JL, Fogley RD, van Rooijen E, Hagedorn EJ, Ciarlo C, White RM, Matos DA, Puller AC, Santoriello C, Liao EC, Young RA, Zon LI. A zebrafish melanoma model reveals emergence of neural crest identity during melanoma initiation. Science. 2016 Jan 29;351(6272):aad2197. doi: 10.1126/science.aad2197. Epub 2016 Jan 28. PubMed PMID: 26823433; PubMed Central PMCID: PMC4868069.
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Wilson NR, Olm-Shipman AJ, Acevedo DS, Palaniyandi K, Hall EG, Kosa E, Stumpff KM, Smith GJ, Pitstick L, Liao EC, Bjork BC, Czirok A, Saadi I. SPECC1L deficiency results in increased adherens junction stability and reduced cranial neural crest cell delamination. Sci Rep. 2016 Jan 20;6:17735. doi: 10.1038/srep17735. PubMed PMID: 26787558; PubMed Central PMCID: PMC4726231.
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Rochard LJ, Ling IT, Kong Y, Liao EC. Visualization of Chondrocyte Intercalation and Directional Proliferation via Zebrabow Clonal Cell Analysis in the Embryonic Meckel's Cartilage. J Vis Exp. 2015 Oct 21;(105):e52935. doi: 10.3791/52935. PubMed PMID: 26555721; PubMed Central PMCID: PMC4692669.
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Gfrerer L, Shubinets V, Hoyos T, Kong Y, Nguyen C, Pietschmann P, Morton CC, Maas RL, Liao EC. Functional analysis of SPECC1L in craniofacial development and oblique facial cleft pathogenesis. Plast Reconstr Surg. 2014 Oct;134(4):748-759. doi: 10.1097/PRS.0000000000000517. PubMed PMID: 25357034; PubMed Central PMCID: PMC4430087.
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Kong Y, Grimaldi M, Curtin E, Dougherty M, Kaufman C, White RM, Zon LI, Liao EC. Neural crest development and craniofacial morphogenesis is coordinated by nitric oxide and histone acetylation. Chem Biol. 2014 Apr 24;21(4):488-501. doi: 10.1016/j.chembiol.2014.02.013. Epub 2014 Mar 27. PubMed PMID: 24684905; PubMed Central PMCID: PMC4349424
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Kinikoglu B, Kong Y, Liao EC. Characterization of cultured multipotent zebrafish neural crest cells. Exp Biol Med (Maywood). 2014 Feb;239(2):159-68. doi: 10.1177/1535370213513997. Epub 2013 Dec 10. PubMed PMID: 24326414.
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Gfrerer L, Dougherty M, Liao EC. Visualization of craniofacial development in the sox10: kaede transgenic zebrafish line using time-lapse confocal microscopy. J Vis Exp. 2013 Sep 30;(79):e50525. doi: 10.3791/50525. PubMed PMID: 24121214; PubMed Central PMCID: PMC3938043.
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Kamel G, Hoyos T, Rochard L, Dougherty M, Kong Y, Tse W, Shubinets V, Grimaldi M, Liao EC. Requirement for frzb and fzd7a in cranial neural crest convergence and extension mechanisms during zebrafish palate and jaw morphogenesis. Dev Biol. 2013 Sep 15;381(2):423-33. doi: 10.1016/j.ydbio.2013.06.012. Epub 2013 Jun 25. PubMed PMID: 23806211.
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Lindgren AM, Hoyos T, Talkowski ME, Hanscom C, Blumenthal I, Chiang C, Ernst C, Pereira S, Ordulu Z, Clericuzio C, Drautz JM, Rosenfeld JA, Shaffer LG, Velsher L, Pynn T, Vermeesch J, Harris DJ, Gusella JF, Liao EC, Morton CC. Haploinsufficiency of KDM6A is associated with severe psychomotor retardation, global growth restriction, seizures and cleft palate. Hum Genet. 2013 May;132(5):537-52. doi: 10.1007/s00439-013-1263-x. Epub 2013 Jan 25. PubMed PMID: 23354975; PubMed Central PMCID: PMC3627823.
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Dougherty M, Kamel G, Grimaldi M, Gfrerer L, Shubinets V, Ethier R, Hickey G, Cornell RA, Liao EC. Distinct requirements for wnt9a and irf6 in extension and integration mechanisms during zebrafish palate morphogenesis. Development. 2013 Jan 1;140(1):76-81. doi: 10.1242/dev.080473. Epub 2012 Nov 15. PubMed PMID: 23154410; PubMed Central PMCID: PMC6514306.
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Dougherty M, Kamel G, Shubinets V, Hickey G, Grimaldi M, Liao EC. Embryonic fate map of first pharyngeal arch structures in the sox10: kaede zebrafish transgenic model. J Craniofac Surg. 2012 Sep;23(5):1333-7. doi: 10.1097/SCS.0b013e318260f20b. PubMed PMID: 22948622.
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Nyame TT, Lemon KP, Kolter R, Liao EC. High-throughput assay for bacterial adhesion on acellular dermal matrices and synthetic surgical materials. Plast Reconstr Surg. 2011 Nov;128(5):1061-1068. doi: 10.1097/PRS.0b013e31822b65af. PubMed PMID: 22030489; PubMed Central PMCID: PMC3766523.
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Curtin E, Hickey G, Kamel G, Davidson AJ, Liao EC. Zebrafish wnt9a is expressed in pharyngeal ectoderm and is required for palate and lower jaw development. Mech Dev. 2011 Jan-Feb;128(1-2):104-15. doi: 10.1016/j.mod.2010.11.003. Epub 2010 Nov 18. PubMed PMID: 21093584.
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Paw BH, Davidson AJ, Zhou Y, Li R, Pratt SJ, Lee C, Trede NS, Brownlie A, Donovan A, Liao EC, Ziai JM, Drejer AH, Guo W, Kim CH, Gwynn B, Peters LL, Chernova MN, Alper SL, Zapata A, Wickramasinghe SN, Lee MJ, Lux SE, Fritz A, Postlethwait JH, Zon LI. Cell-specific mitotic defect and dyserythropoiesis associated with erythroid band 3 deficiency. Nat Genet. 2003 May;34(1):59-64. doi: 10.1038/ng1137. PubMed PMID: 12669066.
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Shafizadeh E, Paw BH, Foott H, Liao EC, Barut BA, Cope JJ, Zon LI, Lin S. Characterization of zebrafish merlot/chablis as non-mammalian vertebrate models for severe congenital anemia due to protein 4.1 deficiency. Development. 2002 Sep;129(18):4359-70. doi: 10.1242/dev.129.18.4359. PubMed PMID: 12183387.
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Liao EC, Trede NS, Ransom D, Zapata A, Kieran M, Zon LI. Non-cell autonomous requirement for the bloodless gene in primitive hematopoiesis of zebrafish. Development. 2002 Feb;129(3):649-59. doi: 10.1242/dev.129.3.649. PubMed PMID: 11830566.
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Liao EC, Paw BH, Peters LL, Zapata A, Pratt SJ, Do CP, Lieschke G, Zon LI. Hereditary spherocytosis in zebrafish riesling illustrates evolution of erythroid beta-spectrin structure, and function in red cell morphogenesis and membrane stability. Development. 2000 Dec;127(23):5123-32. doi: 10.1242/dev.127.23.5123. PubMed PMID: 11060238.
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Oates AC, Brownlie A, Pratt SJ, Irvine DV, Liao EC, Paw BH, Dorian KJ, Johnson SL, Postlethwait JH, Zon LI, Wilks AF. Gene duplication of zebrafish JAK2 homologs is accompanied by divergent embryonic expression patterns: only jak2a is expressed during erythropoiesis. Blood. 1999 Oct 15;94(8):2622-36. PubMed PMID: 10515866.
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Porcher C, Liao EC, Fujiwara Y, Zon LI, Orkin SH. Specification of hematopoietic and vascular development by the bHLH transcription factor SCL without direct DNA binding. Development. 1999 Oct;126(20):4603-15. doi: 10.1242/dev.126.20.4603. PubMed PMID: 10498694.
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Postlethwait JH, Yan YL, Gates MA, Horne S, Amores A, Brownlie A, Donovan A, Egan ES, Force A, Gong Z, Goutel C, Fritz A, Kelsh R, Knapik E, Liao E, Paw B, Ransom D, Singer A, Thomson M, Abduljabbar TS, Yelick P, Beier D, Joly JS, Larhammar D, Rosa F, Westerfield M, Zon LI, Johnson SL, Talbot WS. Vertebrate genome evolution and the zebrafish gene map. Nat Genet. 1998 Apr;18(4):345-9. doi: 10.1038/ng0498-345. PubMed PMID: 9537416.
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Liao EC, Paw BH, Oates AC, Pratt SJ, Postlethwait JH, Zon LI. SCL/Tal-1 transcription factor acts downstream of cloche to specify hematopoietic and vascular progenitors in zebrafish. Genes Dev. 1998 Mar 1;12(5):621-6. doi: 10.1101/gad.12.5.621. PubMed PMID: 9499398; PubMed Central PMCID: PMC316577.