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Preprints Under Review

  • Kostyanovskaya E, Lasser MC, Wang B, Schmidt J, Bader E, Buteo C, Arbelaez J, Sindledecker AR, McCluskey KE, Castillo O, Wang S, Dea J, Helde K,  Graglia JM, Brimble E, Kastner DB, Ehrlich A, State MW, Willsey AJ, Willsey HR. Convergence of autism proteins at the cilium. Preprint (2025). PMID: 39677731 PubMed PDF

  • Schmidt J, Terala A, McCluskey K, Wang B, Pfalzer A, Willsey HR. Koolen-de Vries Syndrome causal gene KANSL1 is required for motile ciliogenesis. Preprint (2024). bioRxiv PDF

  • Teerikorpi N, Lasser M, Wang S, Kostyanovskaya E, Bader E, Sun N, Dea J, Nowakowski T, Willsey AJ, Willsey HR. Ciliary biology intersects autism and congenital heart disease. Preprint (2024). PMID: 39131273 PubMed PDF

  • Sun N, Teyssier N, Wang B, Drake S, Seyler M, Zaltsmnan Y, Everitt A, Teerikorpi N, Willsey HR, Goodarzi H, Tian R, Kampmann M, Willsey AJ. Autism genes converge on microtubule biology and RNA-binding proteins during excitatory neurogenesis. Preprint (2024). PMID: 38187634 PubMed PDF

  • Wang B*, Vartak R*, Zaltsman Y*, Chi Naing ZZ*, Hennick K, Polacco B, Bashir A, Eckhardt M, Bouhaddou M, Xu J, Sun N, Lasser M, Zhou Y, McKetney J, Guiley K, Chan U, Kaye J, Chadha N, Cakir M, Gordon M, Khare P, Drake S, Drury V, Burke D, Gonzalez S, Alkhairy S, Thomas R, Lam S, Morris M, Bader E, Seyler M, Baum T, Krasnoff R, Wang S, Pham P, Arbalaez J, Pratt D, Chag S, Mahmoo N, Rolland T, Bourgeron T, Finkbeiner S, Swaney D, Bandyopadha S, Ideker T, Beltrao P, Willsey HR, Obernier K, Nowakowski TJ, Hüttenhain R, State MW, Willsey AJ, Krogan NJ. A foundational atlas of autism protein interactions reveals molecular convergence. Preprint (2024). *co-first author PMID: 38076945 PubMed PDF

    Peer-Reviewed Articles

  • McCluskey K, Stovell K,  Law K,  Kostyanovskaya E,  Schmidt J, Exner CRT, Dea J, Brimble E, State MW, Willsey AJ, Willsey HR. Autism gene variants disrupt enteric neuron migration and cause gastrointestinal dysmotility. Nature Communications, In Press (2025). PMID: 38854068 PubMed PDF

  • Thiruppathy M, Teubner L, Roberts R, Lasser M, Moscatello A, Chen Y, Hochstim C, Ruffins S, Sarkar A, Tassey J, Evseenko D, Lozito T, Willsey HR, Gillis JA, Crump JG. Repurposing of a gill gene regulatory program for outer ear evolution. Nature (2025). PMID: 39788155 PubMed PDF

  • Willsey HR, Seaby E, Godwin A, Ennis S, Guille M, Grainger R. Modelling human genetic disorders in Xenopus tropicalis. Disease Models and Mechanisms (2024). PMID: 38832520 PubMed PDF

  • Lasser M, Sun N, Xu Y, Law K, Gonzalez S, Wang B, Drury V, Drake S, Zaltsman Y, Dea J, Bader E, McCluskey KE, State MW, Willsey AJ, Willsey HR. Pleiotropy of autism-associated chromatin regulators. Development (2023). PMID: 37366052 PubMed PDF

  • Willsey HR*, Willsey AJ*, Wang B, State MW. Genomics, convergent neuroscience and progress in understanding autism spectrum disorder. Nature Reviews Neuroscience (2022).*co-first author PMID: 35440779 PubMed PDF

  • Willsey HR, Guille M, Grainger RM. Modeling human genetic disorders with CRISPR technologies in Xenopus. Cold Spring Harbor Protocols (2022). Cover Art. PMID: 34531330 PubMed PDF

  • Naert T, Çiçek O, Ogar P, Bürgi M, Shaidani N, Kaminski M, Xu Y, Grand K, Vujanovic M, Prata D, Hildebrandt F, Brox T, Ronneberger O, Voigt FF, Helmchen F, Loffing J, Horb ME, Willsey HR, Lienkamp, SS. Deep learning is widely applicable to phenotyping embryonic development and disease. Development (2021). PMID: 34739029 PubMed PDF

  • Ly VT*, Baudin PV*, Pansodtee P, Jung EA, Voitiuk K, Rosen YM, Willsey HR, Mantalas GL, Seiller ST, Selberg JA, Cordero SA, Ross JM, Rolandi M, Pollen AA, Nowakowski TJ, Haussler D, Mostajo-Radji MA, Salama SR, Teodorescu M. Picroscope: Low-Cost System for Simultaneous Longitudinal Biological Imaging. Communications Biology (2021). *co-first author PMID: 34737378 PubMed PDF

  • Baudin PV*, Ly VT*, Pansodtee P, Jung EA, Currie R, Hoffman R, Willsey HR, Pollen AA, Nowakowski TJ, Haussler D, Mostajo-Radji MA, Salama SR, Teodorescu M. Low cost cloud based remote microscopy for biological sciences. Internet of Things (2021). *co-first author PDF

  • Rosenthal SB*, Willsey HR*, Xu Y, Mei Y, Dea J, Wang S, Curtis C, Sempu E, Khokha MK, Chi NC, Willsey, AJ, Fisch KM, Ideker T. A convergent molecular network underlying autism and congenital heart disease. Cell Systems (2021).*co-first author PMID: 34411509 PubMed PDF

  • Willsey HR. Whole-mount RNA in situ hybridization and immunofluorescence of Xenopus embryos and tadpoles. Cold Spring Harbor Protocols (2021). Cover Art. PMID: 33827967 PubMed PDF

  • Willsey HR, Exner CRT, Xu Y, Everitt A, Dea J, Schmunk G, Sun N, Wang B, Zaltsman Y, Teerikorpi N, Kim A, Anderson AS, Shin D, Seyler M, Nowakowski TJ, Harland RM, Willsey AJ, State MW. Parallel in vivo analysis of large-effect autism genes implicates cortical neurogenesis and estrogen in risk and resilience. Neuron (2021). Featured article. Cover Art. PMID: 33497602 PubMed PDF

  • Willsey HR, Exner CRT. Xenopus leads the way: Frogs as a pioneering model to understand the human brain. Genesis (2020). PMID: 33369095 PubMed PDF

  • Willsey HR*, Xu Y*, Everitt A, Dea J, Exner CRT, Willsey AJ, State MW, Harland RM. The neurodevelopmental disorder risk gene DYRK1A is required for ciliogenesis and control of brain size in Xenopus embryos. Development (2020). Cover Art. *co-first author PMID: 32467234 PubMed PDF

  • Dur AH, Tang T, Viviano S, Sekuri A, Willsey HR, Tagare HD, Kahle KT, Deniz E. In Xenopus ependymal cilia drive embryonic CSF circulation and brain development independently of cardiac pulsatile forces. Fluids Barriers CNS (2020). PMID: 33308296 PubMed PDF

  • Blackburn ATM, Bekheirnia N, Uma VC, Corkins ME, Xu Y, Rosenfeld JA, Bainbridge MN, Yang Y, Liu P, Madan-Khetarpal S, Delgado MR, Hudgins L, Krantz I, Rodriguez-Buritica D, Wheeler PG, Al Gazali L, Al Shamsi AMSM, Gomez-Ospina N, Chao H, Mirzaa GM, Scheuerle AE, Kukolich MK, Scaglia F, Eng C, Willsey HR, Braun MC, Lamb DJ, Miller RK, Bekheirnia MR. DYRK1A-related intellectual disability: a syndrome associated with congenital anomalies of the kidney and urinary tract. Genetics in Medicine (2019). PMID: 31263215 PubMed PDF

  • Willsey HR, Walentek P, Exner CRT, Xu Y, Lane A, Harland RM, Heald R, Santama N. Katanin-like protein Katnal2 is required for ciliogenesis and brain development in Xenopus embryos. Developmental Biology (2018). PMID: 30096282 PubMed PDF

  • Willsey AJ, Morris MT, Wang S, Willsey HR, Sun N, Teerikorpi N, Baum TB, Cagney G, Bender KJ, Desai TA, Srivastava D, Davis GW, Doudna J, Chang E, Sohal V, Lowenstein DH, Li H, Agard D, Keiser MJ, Shoichet B, Zastrow von M, Mucke L, Finkbeiner S, Gan L, Sestan N, Ward ME, Huttenhain R, Nowakowski TJ, Bellen HJ, Frank LM, Khokha MK, Lifton RP, Kampmann M, Ideker T, State MW, Krogan NJ. The Psychiatric Cell Map Initiative: A Convergent Systems Biological Approach to Illuminating Key Molecular Pathways in Neuropsychiatric Disorders. Cell (2018). PMID: 30096282 PubMed PDF

  • Willsey HR, Zheng X, Pastor-Pareja JC, Willsey AJ, Beachy PA, Xu T. Localized JNK signaling regulates organ size during development. eLife (2016). PMID: 26974344 PubMed PDF

  • Leung W.,… Rankin HB,… Spana EP,…Elgin SCR. Drosophila Muller F Elements Maintain a Distinct Set of Genomic Properties Over 40 Million Years of Evolution. G3 (2015). PMID: 25740935 PubMed PDF

  • Maynard JC, Pham T, Zheng T, Jockheck-Clark A, Rankin HB, Newgard CB, Spana EP, Nicchitta CV. Gp93, the Drosophila GRP94 ortholog, is required for gut epithelial homeostasis and nutrient assimilation-coupled growth control. Developmental Biology (2010). PMID: 20044986 PubMed PDF