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  • 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). bioRxiv PDF

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

  • McCluskey K, Stovell K,  Law K,  Kostyanovskaya E,  Schmidt J, Exner CRT, Dea J, Brimble E, State MW, Willsey AJ, Willsey HR. (2024) Autism gene variants disrupt enteric neuron migration and cause gastrointestinal dysmotility. Preprint. bioRxiv 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). bioRxiv 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 (2023). *co-first author bioRxiv 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) 150 (14): PMID: 37366052. PubMed PDF

  • Willsey HR*, Willsey AJ*, Wang B, State MW. Genomics, convergent neuroscience and progress in understanding autism spectrum disorder. Nat Rev Neurosci. 2022 06; 23(6):323-341. PMID: 35440779. *co-first author PubMed PDF

  • Willsey HR, Guille M, Grainger RM. Modeling human genetic disorders with CRISPR technologies in Xenopus. Cold Spring Harbor Protocols. 2022 Mar 1;2022(3). 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 (2021) Deep learning is widely applicable to phenotyping embryonic development and disease. Development 148(21):dev199664. 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 (2021). Picroscope: Low-Cost System for Simultaneous Longitudinal Biological Imaging. Communications Biology 4(1):1261. *co-first author 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 (2021) Low cost cloud based remote microscopy for biological sciences. Internet of Things https://doi.org/10.1016/j.iot.2021.100454. *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 12(11):1094-1107.e6. PMID: 34411509.*co-first author PubMed PDF

  • Willsey HR. Whole-mount RNA in situ hybridization and immunofluorescence of Xenopus embryos and tadpoles. Cold Spring Harbor Protocols. 2021 Oct 1; 2021 (10):pdb.prot105635 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 109(5):788-804. 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 59(1-2):e23405. 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 147(21):dev189290. Cover Art. PMID: 32467234. *co-first author PubMed PDF

  • Dur AH, Tang T, Viviano S, Sekuri A, Willsey HR, Tagare HD, Kahle KT, Deniz E. (2020) In Xenopus ependymal cilia drive embryonic CSF circulation and brain development independently of cardiac pulsatile forces. Fluids Barriers CNS 17(1):72. 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. (2019) DYRK1A-related intellectual disability: a syndrome associated with congenital anomalies of the kidney and urinary tract. Genetics in Medicine 21(12):2755-2764. PubMed PDF

  • Willsey HR, Walentek P, Exner CRT, Xu Y, Lane A, Harland RM, Heald R, Santama N. (2018) Katanin-like protein Katnal2 is required for ciliogenesis and brain development in Xenopus embryos. Developmental Biology 442, 276-287. 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. (2018) The Psychiatric Cell Map Initiative: A Convergent Systems Biological Approach to Illuminating Key Molecular Pathways in Neuropsychiatric Disorders. Cell 174, 505–520. PubMed PDF

  • Willsey HR, Zheng X, Pastor-Pareja JC, Willsey AJ, Beachy PA, Xu T. (2016) Localized JNK signaling regulates organ size during development. eLife 5, 375. PubMed PDF

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

  • Maynard JC, Pham T, Zheng T, Jockheck-Clark A, Rankin HB, Newgard CB, Spana EP, Nicchitta CV. (2010) Gp93, the Drosophila GRP94 ortholog, is required for gut epithelial homeostasis and nutrient assimilation-coupled growth control. Developmental Biology 339, 295–306. PubMed PDF