Publications

2023    2022    2021    2020    2019    2018    2017    2016    2015    2014    2013    2012

2023

Application of an automated analysis framework for pulsed-wave Doppler cardiac ultrasound measurements to generate reference data in adult zebrafish. Van Impe M, Caboor L, Deleeuw V, De Rycke K, Vanhooydonck M, De Backer J, Segers P, Sips P. Am J Physiol Regul Integr Comp Physiol. 2023 Dec 1;325(6):R782-R796. doi: 10.1152/ajpregu.00103.2023. Epub 2023 Oct 9. PMID: 37811715.

Significance of Premature Vertebral Mineralization in Zebrafish Models in Mechanistic and Pharmaceutical Research on Hereditary Multisystem Diseases. Wynsberghe JV, Vanakker OM. Biomolecules. 2023 Nov 6;13(11):1621. doi: 10.3390/biom13111621. PMID: 38002303; PMCID: PMC10669475.

Syntaxin 18 Defects in Human and Zebrafish Unravel Key Roles in Early Cartilage and Bone Development. Guillemyn B, De Saffel H, Bek JW, Tapaneeyaphan P, De Clercq A, Jarayseh T, Debaenst S, Willaert A, De Rycke R, Byers PH, Rosseel T, Coucke P, Blaumeiser B, Syx D, Malfait F, Symoens S. J Bone Miner Res. 2023 Nov;38(11):1718-1730. doi: 10.1002/jbmr.4914. Epub 2023 Sep 30. PMID: 37718532.

Cell differentiation and matrix organization are differentially affected during bone formation in osteogenesis imperfecta zebrafish models with different genetic defects impacting collagen type I structure. Daponte V, Tonelli F, Masiero C, Syx D, Exbrayat-Héritier C, Biggiogera M, Willaert A, Rossi A, Coucke PJ, Ruggiero F, Forlino A. Matrix Biol. 2023 Aug;121:105-126. doi: 10.1016/j.matbio.2023.06.003. Epub 2023 Jun 17. PMID: 37336269.

Poly (A)-specific ribonuclease deficiency impacts oogenesis in zebrafish. Nanjappa DP, De Saffel H, Kalladka K, Arjuna S, Babu N, Prasad K, Sips P, Chakraborty A. Sci Rep. 2023 Jun 20;13(1):10026. doi: 10.1038/s41598-023-37226-6. PMID: 37340076; PMCID: PMC10281955.

Atm deficient zebrafish model reveals conservation of the tumour suppressor function and a role in fertility. Vierstraete J, Fieuws C, Creytens D, Van Dorpe J, Willaert A, Vral A, Claes KB. Genes Dis. 2022 May 13;10(2):381-384. doi: 10.1016/j.gendis.2022.04.023. PMID: 37223540; PMCID: PMC10201593.

A tapt1 knock-out zebrafish line with aberrant lens development and impaired vision models human early-onset cataract. Jarayseh T, Guillemyn B, De Saffel H, Bek JW, Syx D, Symoens S, Gansemans Y, Van Nieuwerburgh F, Jagadeesh S, Raja J, Malfait F, Coucke PJ, De Clercq A, Willaert A. Hum Genet. 2023 Mar;142(3):457-476. doi: 10.1007/s00439-022-02518-w. Epub 2023 Jan 25. PMID: 36697720.

Zebrafish Tric-b is required for skeletal development and bone cells differentiation. Tonelli F, Leoni L, Daponte V, Gioia R, Cotti S, Fiedler IAK, Larianova D, Willaert A, Coucke PJ, Villani S, Busse B, Besio R, Rossi A, Witten PE, Forlino A. Front Endocrinol (Lausanne). 2023 Jan 23;14:1002914. doi: 10.3389/fendo.2023.1002914. PMID: 36755921; PMCID: PMC9899828.

2022

Atm deficient zebrafish model reveals conservation of the tumour suppressor function and a role in fertility. Vierstraete J, Fieuws C, Creytens D, Van Dorpe J, Willaert A, Vral A, Claes KB. Genes Dis. 2022 May 13;10(2):381-384. doi: 10.1016/j.gendis.2022.04.023. PMID: 37223540; PMCID: PMC10201593.

Therapeutic targeting of LCK tyrosine kinase and mTOR signaling in T-cell acute lymphoblastic leukemia. Laukkanen S, Veloso A, Yan C, Oksa L, Alpert EJ, Do D, Hyvärinen N, McCarthy K, Adhikari A, Yang Q, Iyer S, Garcia SP, Pello A, Ruokoranta T, Moisio S, Adhikari S, Yoder JA, Gallagher K, Whelton L, Allen JR, Jin AH, Loontiens S, Heinäniemi M, Kelliher M, Heckman CA, Lohi O, Langenau DM. Blood. 2022 Oct 27;140(17):1891-1906. doi: 10.1182/blood.2021015106. PMID: 35544598; PMCID: PMC10082361.

RRM2 enhances MYCN-driven neuroblastoma formation and acts as a synergistic target with CHK1 inhibition. Nunes C, Depestel L, Mus L, Keller KM, Delhaye L, Louwagie A, Rishfi M, Whale A, Kara N, Andrews SR, Dela Cruz F, You D, Siddiquee A, Cologna CT, De Craemer S, Dolman E, Bartenhagen C, De Vloed F, Sanders E, Eggermont A, Bekaert SL, Van Loocke W, Bek JW, Dewyn G, Loontiens S, Van Isterdael G, Decaesteker B, Tilleman L, Van Nieuwerburgh F, Vermeirssen V, Van Neste C, Ghesquiere B, Goossens S, Eyckerman S, De Preter K, Fischer M, Houseley J, Molenaar J, De Wilde B, Roberts SS, Durinck K, Speleman F. Sci Adv. 2022 Jul 15;8(28):eabn1382. doi: 10.1126/sciadv.abn1382. Epub 2022 Jul 13. PMID: 35857500; PMCID: PMC9278860.

Minocycline Attenuates Excessive DNA Damage Response and Reduces Ectopic Calcification in Pseudoxanthoma Elasticum. Nollet L, Van Gils M, Willaert A, Coucke PJ, Vanakker OM. J Invest Dermatol. 2022 Jun;142(6):1629-1638.e6. doi: 10.1016/j.jid.2021.10.019. Epub 2021 Nov 4. PMID: 34742705.

The Abcc6a Knockout Zebrafish Model as a Novel Tool for Drug Screening for Pseudoxanthoma Elasticum. Van Gils M, Willaert A, Coucke PJ, Vanakker OM. Front Pharmacol. 2022 Mar 4;13:822143. doi: 10.3389/fphar.2022.822143. PMID: 35317004; PMCID: PMC8934400.

Minocycline Counteracts Ectopic Calcification in a Murine Model of Pseudoxanthoma Elasticum: A Proof-of- Concept Study. Bouderlique E, Nollet L, Letavernier E, Vanakker OM. Int J Mol Sci. 2022 Feb 6;23(3):1838. doi: 10.3390/ijms23031838. PMID: 35163765; PMCID: PMC8837001.

2021

Animal Models of Ehlers- Danlos Syndromes: Phenotype, Pathogenesis, and Translational Potential. Vroman R, Malfait AM, Miller RE, Malfait F, Syx D. Front Genet. 2021 Oct 12;12:726474. doi: 10.3389/fgene.2021.726474. PMID: 34712265; PMCID: PMC8547655.

Lrp5 Mutant and Crispant Zebrafish Faithfully Model Human Osteoporosis, Establishing the Zebrafish as a Platform for CRISPR- Based Functional Screening of Osteoporosis Candidate Genes. Bek JW, Shochat C, De Clercq A, De Saffel H, Boel A, Metz J, Rodenburg F, Karasik D, Willaert A, Coucke PJ. J Bone Miner Res. 2021 Sep;36(9):1749-1764. doi: 10.1002/jbmr.4327. Epub 2021 May 19. PMID: 33957005.

Loss of zebrafish atp6v1e1b, encoding a subunit of vacuolar ATPase, recapitulates human ARCL type 2C syndrome and identifies multiple pathobiological signatures. Pottie L, Van Gool W, Vanhooydonck M, Hanisch FG, Goeminne G, Rajkovic A, Coucke P, Sips P, Callewaert B. PLoS Genet. 2021 Jun 18;17(6):e1009603. doi: 10.1371/journal.pgen.1009603. PMID: 34143769; PMCID: PMC8244898.

Bi-allelic premature truncating variants in LTBP1 cause cutis laxa syndrome. Pottie L, Adamo CS, Beyens A, Lütke S, Tapaneeyaphan P, De Clercq A, Salmon PL, De Rycke R, Gezdirici A, Gulec EY, Khan N, Urquhart JE, Newman WG, Metcalfe K, Efthymiou S, Maroofian R, Anwar N, Maqbool S, Rahman F, Altweijri I, Alsaleh M, Abdullah SM, Al-Owain M, Hashem M, Houlden H, Alkuraya FS, Sips P, Sengle G, Callewaert B. Am J Hum Genet. 2021 Jun 3;108(6):1095-1114. doi: 10.1016/j.ajhg.2021.04.016. Epub 2021 May 14. Erratum in: Am J Hum Genet. 2021 Dec 2;108(12):2386-2388. PMID: 33991472; PMCID: PMC8206382.

Photoconvertible fluorescent proteins: a versatile tool in zebrafish skeletal imaging. Bek JW, De Clercq A, De Saffel H, Soenens M, Huysseune A, Witten PE, Coucke PJ, Willaert A. J Fish Biol. 2021 Apr;98(4):1007-1017. doi: 10.1111/jfb.14335. Epub 2020 May 8. PMID: 32242924.

PRL3 enhances T-cell acute lymphoblastic leukemia growth through suppressing T-cell signaling pathways and apoptosis. Leukemia. Garcia EG, Veloso A, Oliveira ML, Allen JR, Loontiens S, Brunson D, Do D, Yan C, Morris R, Iyer S, Garcia SP, Iftimia N, Van Loocke W, Matthijssens F, McCarthy K, Barata JT, Speleman F, Taghon T, Gutierrez A, Van Vlierberghe P, Haas W, Blackburn JS, Langenau DM. 2021 Mar;35(3):679-690. doi: 10.1038/s41375-020-0937-3. Epub 2020 Jun 30. PMID: 32606318; PMCID: PMC8009053.

The ZE-Tunnel: An Affordable, Easy-to-Assemble, and User-Friendly Benchtop Zebrafish Swim Tunnel. Bek JW, De Clercq A, Coucke PJ, Willaert A. Zebrafish. 2021 Feb;18(1):29-41. doi: 10.1089/zeb.2020.1948. Epub 2021 Jan 12. PMID: 33428527.

Zebrafish as an in vivo screening tool to establish PARP inhibitor efficacy. Vierstraete J, Fieuws C, Willaert A, Vral A, Claes KBM. DNA Repair (Amst). 2021 Jan;97:103023. doi: 10.1016/j.dnarep.2020.103023. Epub 2020 Nov 12. PMID: 33341473.

2020

Delbaere S, De Clercq A, Mizumoto S, Noborn F, Bek JW, Alluyn L, Gistelinck C, Syx D, Salmon PL, Coucke PJ, Larson G, Yamada S, Willaert A and Malfait F (2020) b3galt6 Knock-Out Zebrafish Recapitulate β3GalT6-Deficiency Disorders in Human and Reveal a Trisaccharide Proteoglycan Linkage Region. Front. Cell Dev. Biol. 8:597857. doi: 10.3389/fcell.2020.597857

Loontiens, S., Vanhauwaert, S., Depestel, L. et al. A novel TLX1-driven T-ALL zebrafish model: comparative genomic analysis with other leukemia models. Leukemia (2020). https://doi.org/10.1038/s41375-020-0938-2

Garcia, E.G., Veloso, A., Oliveira, M.L. et al. PRL3 enhances T-cell acute lymphoblastic leukemia growth through suppressing T-cell signaling pathways and apoptosis. Leukemia (2020). https://doi.org/10.1038/s41375-020-0937-3

2019

S. Delbaere, T. Van Damme, D. Syx, et al., Hypomorphic zebrafish models mimic the musculoskeletalphenotype ofb4GalT7-deficient Ehlers-Danlos syndr..., Matrix Biology,https://doi.org/10.1016/j.matbio.2019.12.002

Van Gils M, Vanakker OM. Morpholino-Mediated Gene Knockdown in Zebrafish: It Is All About Dosage and Validation. J Invest Dermatol. 2019 Jul;139(7):1599-1600.

Oralová V, Rosa JT, Soenens M, Bek JW, Willaert A, Witten PE, Huysseune A. Beyond the whole-mount phenotype: high-resolution imaging in fluorescence-based applications on zebrafish. Biol Open. 2019 May 24;8(5).

Loontiens S, Depestel L, Vanhauwaert S, Dewyn G, Gistelinck C, Verboom K, Van Loocke W, et al. Purification of High-Quality Rna from a Small Number of Fluorescence Activated Cell Sorted Zebrafish Cells for Rna Sequencing Purposes. BMC Genomics. 2019 Mar 20;20(1):228.

Cayuela ML, Claes KBM, Ferreira MG, Henriques CM, van Eeden F, Varga M,7, Vierstraete J, Mione MC. The Zebrafish as an Emerging Model to Study DNA Damage in Aging, Cancer and Other Diseases. Front Cell Dev Biol. 2019 Jan 10;6:178.

2018

Boel A, De Saffel H, Steyaert W, Callewaert B, De Paepe A, Coucke PJ, Willaert A. CRISPR/Cas9-mediated homology-directed repair by ssODNs in zebrafish induces complex mutational patterns resulting from genomic integration of repair-template fragments. Dis Model Mech. 2018 Oct 18;11(10).

Steyaert W, Boel A, Coucke P and Willaert A. BATCH-GE: Analysis of NGS Data for Genome Editing Assessment. Methods Mol Biol. 2018;1865:83-90. doi: 10.1007/978-1-4939-8784-9_6.

Gistelinck C, Kwon RY, Malfait F, Symoens S, Harris MP, Henke K, Hawkins MB, Fisher S, Sips P, Guillemyn B, Bek JW, Vermassen P, De Saffel H, Witten PE, Weis M, De Paepe A, Eyre DR, Willaert A*, Coucke PJ*. Zebrafish type I collagen mutants faithfully recapitulate human type I collagenopathies. Proc Natl Acad Sci U S A. 2018 Aug 21;115(34):E8037-E8046. *These authors jointly supervised this work.

Garcia EG, Iyer S, Garcia SP, Loontiens S, Sadreyev RI, Speleman F, Langenau DM. Cell of origin dictates aggression and stem cell number in acute lymphoblastic leukemia. Leukemia. 2018 Aug;32(8):1860-1865

2017

Van Gils M, Willaert A, De Vilder EYG, Coucke PJ, Vanakker OM. Generation and Validation of a Complete Knockout Model of abcc6a in Zebrafish. J Invest Dermatol. 2018 Nov;138(11):2333-2342. doi: 10.1016/j.jid.2018.06.183. Epub 2018 Jul 17.

Hur M, Gistelinck CA, Huber P, Lee J, Thompson MH, Monstad-Rios AT, Watson CJ, McMenamin SK, Willaert A, Parichy DM, Coucke P, Kwon RY. "microCT-based phenomics in the zebrafish skeleton reveals virtues of deep phenotyping in a distributed organ system. Zebrafish. 2017 Nov 27. doi: 10.1089/zeb.2017.1540.

Vierstraete J, Willaert A, Vermassen P, Coucke PJ, Vral A, Claes KBM. Accurate quantification of homologous recombination in zebrafish: brca2 deficiency as a paradigm. Sci Rep. 2017 Nov 28;7(1):16518.

Hur M, Gistelinck CA, Huber P, Lee J, Thompson MH, Monstad-Rios AT, Watson CJ, McMenamin SK, Willaert A, Parichy DM, Coucke P, Kwon RY. "microCT-based phenomics in the zebrafish skeleton reveals virtues of deep phenotyping in a distributed organ system. Elife. 2017 Sep 8;6. pii: e26014. doi: 10.7554/eLife.26014.

2016

Gistelinck C, Eckhard Witten P, Huysseune A, Symoens S, Malfait F, Larionova D, Simoens P, Dierick M, Van Hoorebeke L, De Paepe A, Kwon RY, Weis M, Eyre DR, Willaert A*, Coucke PJ*. Loss of Type I Collagen Telopeptide Lysyl Hydroxylation Causes Musculoskeletal Abnormalities in a Zebrafish Model of Bruck Syndrome. J Bone Miner Res. 2016 Aug 19. doi: 10.1002/jbmr.2977. *These authors jointly supervised this work.

Boel A, Steyaert W, De Rocker N, Menten B, Callewaert B, De Paepe A, Coucke P, Willaert A. BATCH-GE: Batch analysis of Next-Generation Sequencing data for genome editing assessment. Sci Rep. 2016 Jul 27;6:30330.

Vanhauwaert S, Lefever S, Coucke P, Speleman F, De Paepe A, Vandesompele J,Willaert A. RT-qPCR gene expression analysis in zebrafish: Preanalytical precautions and use of expressed repetitive elements for normalization. Methods Cell Biol. 2016;135:329-42. (IF:1,097)

C. Gistelinck*, R. Gioia*, A. Gagliardi, F. Tonelli, L. Marchese, L. Bianchi, C. Landi, L. Bini, A. Huysseune, P. E. Witten, A. Staes, K. Gevaert, N. De Rocker, B. Menten, F. Malfait, S. Leikin, S. Carra, R. Tenni, A. Rossi, A. De Paepe, P. Coucke, A. Willaert†, A. Forlino†. Zebrafish Collagen Type I: Molecular and Biochemical Characterization of the Major Structural Protein in Bone and Skin. Sci Rep. 2016 Feb 15;6:21540). *These authors contributed equally to this work. *These authors jointly supervised this work.

2015

Sofie Symoens, Aileen M. Barnes, Charlotte Gistelinck, Fransiska Malfait, Brecht Guillemyn, Wouter Steyaert, Delfien Syx, Sanne D’hondt, Martine Biervliet, Julie De Backer, Eckhard P. Witten, Sergey Leikin, Elena Makareeva, Gabriele Gillessen-Kaesback, Ann Huysseune, Kris Vleminckx, Andy Willaert, Anne De Paepe, Joan C. Marini, Paul J. Coucke. Genetic defects in TAPT1 disrupt ciliogenesis and cause an in utero lethal osteochondrodysplasia. Am J Hum Genet. 2015 Oct 1;97(4):521-34.

Nina De Rocker, Sarah Vergult, Eva Jacobs, Susan Zeesman, Frédérique Béna, Nele Bockaert, Thomy de Ravel, Koenraad Devriendt, Sabrina Giglio, Laurence Faivre, Shelagh Joss, David Koolen, Saskia Maas, Ernie M Bongers, Francesca Novara, Malgorzata JM Nowaczyk, Hilde Peeters, Abeltje Polstra, Filip Roelens, Carla Rosenberg, Julien Thevenon, Zeynep Tümer, Suzanne Vanhauwaert, Konstantinos Varvagiannis, Andy Willaert, Marjolein Willemsen, Marjolaine Willems, Orsetta Zuffardi, Paul Coucke, Frank Speleman, Tjitske Kleefstra, Björn Menten. Refinement of the critical 2p25.3 deletion region: the role of MYT1L in intellectual disability and obesity. Genet Med. 2015 Jun;17(6):460-6.

2014

Suzanne Vanhauwaert, Gert Van Peer, Ali Rihani, Els Janssens, Pieter Rondou, Steve Lefever, Anne De Paepe, Paul J. Coucke, Frank Speleman, Jo Vandesompele, Andy Willaert. Expressed repeat elements improve RT-qPCR normalization across a wide range of zebrafish gene expression studies. PlosOne 2014 Oct 13;9(10):e109091

Vulto-van Silfhout AT, Rajamanickam S, Jensik PJ, Vergult S, de Rocker N, Newhall KJ, Raghavan R, Reardon SN, Jarrett K, McIntyre T, Bulinski J, Ownby SL, Huggenvik JI, McKnight GS, Rose GM, Cai X, Willaert A, Zweier C, Endele S, de Ligt J, van Bon BW, Lugtenberg D, de Vries PF, Veltman JA, van Bokhoven H, Brunner HG, Rauch A, de Brouwer AP, Carvill GL, Hoischen A, Mefford HC, Eichler EE, Vissers LE, Menten B, Collard MW, de Vries BB. Mutations Affecting the SAND Domain of DEAF1 Cause Intellectual Disability with Severe Speech Impairment and Behavioral Problems. Am J Hum Genet. 2014 May 1;94(5):649-61.

2013

Hosen MJ, Vanakker OM, Willaert A, Huysseune A, Coucke P, De Paepe A Zebrafish models for ectopic mineralization disorders: practical issues from morpholino design to post-injection observations. Front Genet. 2013 May 8;4:74.

2012

Willaert A,* Khatri S,* Callewaert B, Coucke P, Crosby S, Lee J, Davis E, Shiva S, Tsang M, De Paepe A, Urban Z. GLUT10 is required for the development of the cardiovascular system and the notochord and connects mitochondrial function to TGFβ signaling. Hum Mol Genet. 2012 15;21(6):1248-59. *These authors equally contributed to this work.