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EN1 Regulates Cell Growth and Proliferation in Human Glioma Cells via Hedgehog Signaling

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PTPRA Phosphatase Regulates GDNF-Dependent RET Signaling and Inhibits the RET Mutant MEN2A Oncogenic Potential

Yadav, L., Pietilä, E. A., Öhman, T., Liu, X., Mahato, A. K., Sidorova, Y. A., Lehti, K., Saarma, M., & Varjosalo, M. T. (2020) · iScience · doi:10.1016/j.isci.2020.100871 · Cited by 21 · see citing works

Altered mechanisms of genital development identified through integration of DNA methylation and genomic measures in hypospadias

Richard, M. A., Sok, P., Canon, S., Nembhard, W. N., Brown, A. L., Peckham‐Gregory, E. C., Ton, M., Ehli, E. A., Kallsen, N. A., Peyton, S., et al. (2020) · Scientific Reports · doi:10.1038/s41598-020-69725-1 · Cited by 21 · see citing works

Cilia, tubby mice, and obesity

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G‐protein‐coupled receptors and localized signaling in the primary cilium during ventral neural tube patterning

Hwang, S., & Mukhopadhyay, S. (2015) · Birth Defects Research Part A: Clinical and Molecular Teratology · doi:10.1002/bdra.23267 · Cited by 20 · see citing works

Chronophin regulates active vitamin B6 levels and transcriptomic features of glioblastoma cell lines cultured under non-adherent, serum-free conditions

Schulze, M., Hutterer, M., Sabo, A., Hoja, S., Lorenz, J., Rothhammer-Hampl, T., Herold‐Mende, C. C., Floßbach, L., Monoranu, C., & Riemenschneider, M. J. (2018) · BMC Cancer · doi:10.1186/s12885-018-4440-4 · Cited by 19 · see citing works

Selective targeting of mu opioid receptors to primary cilia

Fagan, R. R., Lee, D. F., Geron, M., Scherrer, G., von Zastrow, M., & Ehrlich, A. T. (2024) · Cell Reports · doi:10.1016/j.celrep.2024.114164 · Cited by 18 · see citing works

Differential Roles of Tubby Family Proteins in Ciliary Formation and Trafficking

Hong, J. J., Kim, K. E., Park, S. Y., Bok, J., Seo, J. T., & Moon, S. J. (2021) · Molecules and Cells · doi:10.14348/molcells.2021.0082 · Cited by 17 · see citing works

A pathogenic variant of TULP3 causes renal and hepatic fibrocystic disease

Khamirani, H. J., Palicharla, V. R., Dastgheib, S. A., Dianatpour, M., Imanieh, M. H., Tabei, S. S., Besse, W., Mukhopadhyay, S., & Liem, K. F. (2022) · Frontiers in Genetics · doi:10.3389/fgene.2022.1021037 · Cited by 17 · see citing works

Molecular and structural perspectives on protein trafficking to the primary cilium membrane

Palicharla, V. R., & Mukhopadhyay, S. (2024) · Biochemical Society Transactions · doi:10.1042/bst20231403 · Cited by 16 · see citing works

Permanent cilia loss during cerebellar granule cell neurogenesis involves withdrawal of cilia maintenance and centriole capping

Constable, S., Ott, C. M., Lemire, A. L., White, K., Xun, Y., Lim, A., Lippincott‐Schwartz, J., & Mukhopadhyay, S. (2024) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.2408083121 · Cited by 16 · see citing works

G‐protein‐coupled receptor signaling and neural tube closure defects

Shimada, I. S., & Mukhopadhyay, S. (2016) · Birth Defects Research · doi:10.1002/bdra.23567 · Cited by 16 · see citing works

Identification of a heterogeneous and dynamic ciliome during embryonic development and cell differentiation

Elliott, K. H., Balchand, S. K., Paese, C. L. B., Chang, C., Yang, Y., Brown, K. M., Rasicci, D. T., He, H., Thorner, K., Chaturvedi, P., et al. (2023) · Development · doi:10.1242/dev.201237 · Cited by 14 · see citing works

TULP3: A potential biomarker in colorectal cancer?

Sartor, I. T. S., Recamonde‐Mendoza, M., & Ashton‐Prolla, P. (2019) · PLoS ONE · doi:10.1371/journal.pone.0210762 · Cited by 14 · see citing works

Ciliary ARL13B inhibits developmental kidney cystogenesis in mouse

Sciver, R. E. V., Long, A. B., Katz, H., Gigante, E. D., & Caspary, T. (2023) · Developmental Biology · doi:10.1016/j.ydbio.2023.05.004 · Cited by 11 · see citing works

Mutations in Ciliary Trafficking Genes affect Sonic Hedgehog-dependent Neural Tube Patterning Differentially along the Anterior–Posterior Axis

Legué, E., & Liem, K. F. (2020) · Neuroscience · doi:10.1016/j.neuroscience.2020.07.015 · Cited by 11 · see citing works

Convergent Degenerated Regulatory Elements Associated with Limb Loss in Limbless Amphibians and Reptiles

Zhu, C., Li, S., Zhang, D.-Z., Zhang, J.-J., Wang, G., Zhou, B., Zheng, J., Xu, W., Wang, Z., Gao, X., et al. (2024) · Molecular Biology and Evolution · doi:10.1093/molbev/msae239 · Cited by 10 · see citing works

Embryo-Induced Changes in the Protein Profile of Bovine Oviductal Extracellular Vesicles

Mazzarella, R., Sánchez, J. M., Fernández-Fuertes, B., Egido, S. G., McDonald, M., Alvarez, A. M., González, E., Falcón‐Pérez, J. M., Azkargorta, M., Elortza, F., et al. (2025) · Molecular & Cellular Proteomics · doi:10.1016/j.mcpro.2025.100935 · Cited by 10 · see citing works

Analysis of Hedgehog Signaling in Mouse Intraflagellar Transport Mutants

Ko, H. W., Liu, A., & Eggenschwiler, J. T. (2009) · Methods in cell biology · doi:10.1016/s0091-679x(08)93017-x · Cited by 8 · see citing works

A defined tubby domain β-barrel surface region of TULP3 mediates ciliary trafficking of diverse cargoes

Palicharla, V. R., Badgandi, H., Hwang, S.-H., Legué, E., Liem, K. F., & Mukhopadhyay, S. (2024) · Molecular Biology of the Cell · doi:10.1091/mbc.e24-09-0426 · Cited by 7 · see citing works

Rare variants in TULP3 abolish the suppressive effect on sonic hedgehog signaling and contribute to human neural tube defects

Kuang, L., Jiang, Y., Chen, S., Su, K., Peng, R., Yang, X., & Wang, H. (2021) · Genes & Diseases · doi:10.1016/j.gendis.2021.11.010 · Cited by 7 · see citing works

TULP3 silencing suppresses cell proliferation, migration and invasion in gastric cancer via the PTEN/Akt/Snail pathway

Song, J. W., Fu, Q., Liu, G., Zhang, C., Wang, Y., Tao, S., Liu, R., & Li, Z. (2022) · Cancer Treatment and Research Communications · doi:10.1016/j.ctarc.2022.100551 · Cited by 7 · see citing works

Deficiency of Wdr60 and Wdr34 cause distinct neural tube malformation phenotypes in early embryos

Lu, Y., Yin, H., Mi, Y., Wu, Y., & Zheng, Y. (2023) · Frontiers in Cell and Developmental Biology · doi:10.3389/fcell.2023.1084245 · Cited by 6 · see citing works

Ciliopathy-Associated Missense Mutations in IFT140 are Tolerated by the Inherent Resilience of the IFT Machinery

Beyer, T., Diwan, G. D., Leonhard, T., Dahlke, K., Klose, F., Stehle, I. F., Seda, M., Bolz, S., Woerz, F., Russell, R. B., et al. (2025) · Molecular &amp; Cellular Proteomics · doi:10.1016/j.mcpro.2025.100916 · Cited by 6 · see citing works

Molecular analyses of the tubby-like protein gene family and their response to salt and high temperature in the foxtail millet (Setaria italica)

Jiang, Z., Chen, X., Ruan, L., Yan, X., & Li, K. (2024) · Functional & Integrative Genomics · doi:10.1007/s10142-024-01458-0 · Cited by 4 · see citing works

Biallelic pathogenic TULP3 variants presenting as neonatal cholestasis, liver fibrosis and neurological manifestations

Li, J.-Q., Li, Y., He, R., Lin, Z., Feng, J., Yang, B. J., Shan, Q.-W., Chen, S., Ye, C., Xing, Q., et al. (2025) · Journal of Medical Genetics · doi:10.1136/jmg-2025-110658 · Cited by 4 · see citing works

Primary Cilia, Sonic Hedgehog Signaling, and Spinal Cord Development

Mariani, L. E., & Caspary, T. (2012) · Springer eBooks · doi:10.1007/978-94-007-5808-7_2 · Cited by 4 · see citing works

Regulatory role of primary cilia in oral and maxillofacial development and disease

Liu, Z., Sa, G., Zhang, Z., Wu, Q., Zhou, J., & Yang, X. (2024) · Tissue and Cell · doi:10.1016/j.tice.2024.102389 · Cited by 4 · see citing works

Pleiotropic patterning response to activation of Shh signaling in the limb apical ectodermal ridge

Wang, C. L., Tsugane, M. H., Scranton, V. L., Kosher, R. A., Pierro, L. J., Upholt, W. B., & Dealy, C. N. (2011) · Developmental Dynamics · doi:10.1002/dvdy.22628 · Cited by 3 · see citing works

Tulp3 deficiency results in ciliopathy phenotypes during zebrafish embryogenesis

Epting, D., Devane, J., Mertes, R., Kayser, S., Helmstädter, M., Metzger, P., Boerries, M., Bergmann, C., & Ott, E. B. (2025) · Scientific Reports · doi:10.1038/s41598-025-16584-3 · Cited by 2 · see citing works

Hedgehog signaling: mechanisms and evolution

Ye, X., & Liu, A. (2011) · Frontiers in Biology · doi:10.1007/s11515-011-1146-2 · Cited by 2 · see citing works

Size‐Modulated Mesoderm‐Endoderm Divergence and Myocardial Cavitation in Micropatterned Cardioids

Hoang, P., McKellar, D. W., Kowalczewski, A. C., Mai Nhu, Y., Chai, M., Lian, X. L., Zheng, Y., Amack, J. D., Tucker, N. R., Vlaminck, I. D., et al. (2026) · Advanced Science · doi:10.1002/advs.202515661 · Cited by 1 · see citing works

Neuronal Cilia and Obesity

Berbari, N. F., Pasek, R. C., & Yoder, B. K. (2012) · Springer eBooks · doi:10.1007/978-94-007-5808-7_6 · Cited by 1 · see citing works

The Joubert gene TMEM67 is required for the correct establishment of spinal dorsal identities in human organoidsPreprint

Wiegering, A., Bools, K., Anselme, I., Metayer-Derout, L., Mercey, O., Balissat, E., Bijek, Y., Catala, M., Schneider‐Maunoury, S., & Stedman, A. (2026) · bioRxiv (Cold Spring Harbor Laboratory) · doi:10.64898/2026.01.20.700675 · Cited by 1 · see citing works

Establishment of a bipartite ciliary signaling compartment in a C. elegans thermosensory neuron

Nguyen, T. P. A. (2014) · Summit (Simon Fraser University)

Identification of TULP3 as a negative regulator of Hedgehog signalling in the mouse

Cameron, D. P. (2015) · QSpace (Queen's University Library)

Studying Hedgehog Signaling During Mouse Neural Tube Development

Constable, S., White, K., Somatilaka, B. N., & Mukhopadhyay, S. (2021) · Methods in molecular biology · doi:10.1007/978-1-0716-1701-4_6

Faculty Opinions recommendation of Variable expressivity of ciliopathy neurological phenotypes that encompass Meckel-Gruber syndrome and Joubert syndrome is caused by complex de-regulated ciliogenesis, Shh and Wnt signalling defects.

Stottmann, R. W. (2013) · Faculty Opinions – Post-Publication Peer Review of the Biomedical Literature · doi:10.3410/f.717988867.793472760

TULP3 NLS inhibition: anin silicostudy to hamper cargo transport to nucleus

Mateen, R. M., Tariq, A., Afzal, M. S., Ali, M., Tipu, I., Hussain, M., Saleem, M., & Naveed, M. (2022) · Journal of Biomolecular Structure and Dynamics · doi:10.1080/07391102.2022.2070283

Deciphering the molecular mechanism associated with the interaction of TULP3 and Histone deacetylases, SIRT1 and SIRT2Preprint

Manjula, R., Krishnappa, G., Sriram, V., & Padmanabhan, B. (2024) · bioRxiv (Cold Spring Harbor Laboratory) · doi:10.1101/2024.12.23.630205

Tulp3 quantitative alleles titrate requirements for viability, brain development, and kidney homeostasis but do not suppress Zfp423 mutations in mice

McCoy, C. A., Concepcion, D., Mezody, M. G., Lara, R. Z., Deshpande, O., Liang, C., Long, R. R., & Hamilton, B. A. (2025) · PLoS Genetics · doi:10.1371/journal.pgen.1011911

Tulp3 is a critical repressor of mouse hedgehog signaling

Cameron, D. A., Pennimpede, T., & Petkovich, M. P. (2009) · Developmental Dynamics · doi:10.1002/dvdy.21926 · Cited by 49 · see citing works

Mouse hitchhiker mutants have spina bifida, dorso-ventral patterning defects and polydactyly: identification of Tulp3 as a novel negative regulator of the Sonic hedgehog pathway

Patterson, V. L., Damrau, C., Paudyal, A., Reeve, B., Grimes, D. T., Stewart, M. E., Williams, D. J., Siggers, P., Greenfield, A., & Murdoch, J. N. (2009) · Human Molecular Genetics · doi:10.1093/hmg/ddp075 · Cited by 101 · see citing works

Identifying the molecular function of Tulp3 in neural tube closure and patterning

Hakim-Rad, K., Patterson, V. L., Paudyal, A., & Murdoch, J. N. (2012) · Cilia · doi:10.1186/2046-2530-1-s1-p69

Tubby-like 3 (TULP3) and mammalian Sonic Hedgehog signaling

Eggenschwiler, J. T., & Norman, R. X. (2009) · Developmental Biology · doi:10.1016/j.ydbio.2009.05.371

Identification of TULP3 as a negative regulator of Hedgehog signalling in the mouse

Cameron, D. P. (2015) · QSpace (Queen's University Library)