Publications

Tissue-Engineered Disease Modeling of Lymphangioleiomyomatosis Exposes a Therapeutic Vulnerability to HDAC Inhibition.
\Pietrobon A, Yockell-Lelièvre J, Melong N, Smith LJ, Delaney SP, Azzam N, Xue C, Merwin N, Lian E, Camacho-Magallanes A, Doré C, Musso G, Julian LM, Kristof AS, Tam RY, Berman JN, Shoichet MS, Stanford WL.

Adv Sci (Weinh). 2023 Jul 3:e2302611. doi: 10.1002/advs.202302611.

phox2ba: The Potential Genetic Link behind the Overlap in the Symptomatology between CHARGE and Central Congenital Hypoventilation Syndromes.
MacLean JE, Wertman JN, Prykhozhij SV, Chedrawe E, Langley S, Steele SL, Ban K, Blake K, Berman JN.

Genes (Basel). 2023 May 15;14(5):1086. doi: 10.3390/genes14051086.

Back to the future: evolutionary biology reveals a key regulatory switch in neuroblastoma pathogenesis.
Wertman JN, Berman JN.

J Clin Invest. 2023 May 15;133(10):e167824. doi: 10.1172/JCI167824.

S1P1 Threonine 236 Phosphorylation Mediates the Invasiveness of Triple-Negative Breast Cancer and Sensitivity to FTY720.
Laroche FJF, Li S, Shen N, Hwang SK, Nguyen G, Yu W, Wong CK, Quinton RJ, Berman JN, Liu CT, Singh A, Ganem NJ, Thiagalingam S, Feng H.

Cells. 2023 Mar 23;12(7):980. doi: 10.3390/cells12070980.

Loss of calpain3b in Zebrafish, a Model of Limb-Girdle Muscular Dystrophy, Increases Susceptibility to Muscle Defects Due to Elevated Muscle Activity.
Prykhozhij SV, Caceres L, Ban K, Cordeiro-Santanach A, Nagaraju K, Hoffman EP, Berman JN.

Genes (Basel). 2023 Feb 15;14(2):492. doi: 10.3390/genes14020492.

Human JAK1 gain of function causes dysregulated myelopoeisis and severe allergic inflammation.
Biggs CM, Cordeiro-Santanach A, Prykhozhij SV, Deveau AP, Lin Y, Del Bel KL, Orben F, Ragotte RJ, Saferali A, Mostafavi S, Dinh L, Dai D, Weinacht KG, Dobbs K, Ott de Bruin L, Sharma M, Tsai K, Priatel JJ, Schreiber RA, Rozmus J, Hosking MC, Shopsowitz KE, McKinnon ML, Vercauteren S, Seear M, Notarangelo LD, Lynn FC, Berman JN, Turvey SE.

JCI Insight. 2022 Dec 22;7(24):e150849. doi: 10.1172/jci.insight.150849.

Pilot study of jadomycin B pharmacokinetics and anti-tumoral effects in zebrafish larvae and mouse breast cancer xenograft models.
McKeown BT, Relja NJ, Hall SR, Gebremeskel S, MacLeod JM, Veinotte CJ, Bennett LG, Ohlund LB, Sleno L, Jakeman DL, Berman JN, Johnston B, Goralski KB.

Can J Physiol Pharmacol. 2022 Nov 1;100(11):1065-1076. doi: 10.1139/cjpp-2022-0152.

Zebrafish models of inflammation in hematopoietic development and disease.
Ketharnathan S, Rajan V, Prykhozhij SV, Berman JN.

Front Cell Dev Biol. 2022 Jul 18;10:955658. doi: 10.3389/fcell.2022.955658.

Low-Dose Metronomic Topotecan and Pazopanib (TOPAZ) in Children with Relapsed or Refractory Solid Tumors: A C17 Canadian Phase I Clinical Trial.
Manji A, Samson Y, Deyell RJ, Johnston DL, Lewis VA, Zorzi AP, Berman JN, Brodeur-Robb K, Morrison E, Kee L, Kumar S, Baruchel S, Whitlock JA, Morgenstern DA.

Cancers (Basel). 2022 Jun 17;14(12):2985. doi: 10.3390/cancers14122985.

KIT D816V is dimerization-independent and activates downstream pathways frequently perturbed in mastocytosis.
Rajan V, Prykhozhij SV, Pandey A, Cohen AM, Rainey JK, Berman JN.

Br J Haematol. 2022 Mar 4. doi: 10.1111/bjh.18116.

Radiation dose enhancement using gold nanoparticles with a diamond linear accelerator target: a multiple cell type analysis.
Piccolo O, Lincoln JD, Melong N, Orr BC, Fernandez NR, Borsavage J, Berman JN, Robar J, Ha MN.

Sci Rep. 2022 Jan 28;12(1):1559. doi: 10.1038/s41598-022-05339-z.

High-dose AraC is essential for the treatment of ML-DS independent of postinduction MRD: results of the COG AAML1531 trial.
Hitzler J, Alonzo T, Gerbing R, Beckman A, Hirsch B, Raimondi S, Chisholm K, Viola S, Brodersen L, Loken M, Tong S, Druley T, O'Brien M, Hijiya N, Heerema-McKenney A, Wang YC, Schore R, Taub J, Gamis A, Kolb EA, Berman JN.

Blood. 2021 Dec 9;138(23):2337-2346. doi: 10.1182/blood.2021012206.

CRISPR Knock-in Designer: Automatic Oligonucleotide Design Software to Introduce Point Mutations by Gene Editing Methods
Sergey V. Prykhozhij, Vinothkumar Rajan, Kevin Ban, and Jason N. Berman

Re:GEN Open. Vol. 1, No. 1. 22 Sep 2021. doi: 10.1089/regen.2021.0025

Stress hematopoiesis induces a proliferative advantage in TET2 deficiency.
Rajan V, Collett K, Woodside R, Prykhozhij SV, Moksa M, Carles A, Wong M, Liebman M, Hirst M, Berman JN.

Leukemia. 2021 Sep 30. doi: 10.1038/s41375-021-01427-7.

STAG2 loss rewires oncogenic and developmental programs to promote metastasis in Ewing sarcoma.
Adane B, Alexe G, Seong BKA, Lu D, Hwang EE, Hnisz D, Lareau CA, Ross L, Lin S, Dela Cruz FS, Richardson M, Weintraub AS, Wang S, Iniguez AB, Dharia NV, Conway AS, Robichaud AL, Tanenbaum B, Krill-Burger JM, Vazquez F, Schenone M, Berman JN, Kung AL, Carr SA, Aryee MJ, Young RA, Crompton BD, Stegmaier K.

Cancer Cell. 2021 Jun 14;39(6):827-844.e10. doi: 10.1016/j.ccell.2021.05.007.

Zebrafish Cancer Predisposition Models.
Kobar K, Collett K, Prykhozhij SV, Berman JN.

Front Cell Dev Biol. 2021 Apr 27;9:660069. doi: 10.3389/fcell.2021.660069. 

The identification of dual protective agents against cisplatin-induced oto- and nephrotoxicity using the zebrafish model
Wertman JN, Melong N, Stoyek MR, Piccolo O, Langley S, Orr B, Steele SL, Razaghi B, Berman JN

Elife. 2020 Jul 28;9:e56235. doi: 10.7554/eLife.56235.

The Canadian Rare Diseases Models and Mechanisms (RDMM) Network: Connecting Understudied Genes to Model Organisms.
Boycott KM, Campeau PM, Howley HE, Pavlidis P, Rogic S, Oriel C, Berman JN, Hamilton RM, Hicks GG, Lipshitz HD, Masson JY, Shoubridge EA, Junker A, Leroux MR, McMaster CR, Michaud JL, Turvey SE, Dyment D, Innes AM, van Karnebeek CD, Lehman A, Cohn RD, MacDonald IM, Rachubinski RA, Frosk P, Vandersteen A, Wozniak RW, Pena IA, Wen XY, Lacaze-Masmonteil T, Rankin C, Hieter P.

Am J Hum Genet. 2020 Feb 6;106(2):143-152. doi: 10.1016/j.ajhg.2020.01.009.

Pathologic Features of Down Syndrome Myelodysplastic Syndrome and Acute Myeloid Leukemia: A Report From the Children's Oncology Group Protocol AAML0431
Mast KJ, Taub JW, Alonzo TA, Gamis AS, Mosse CA, Mathew P, Berman JN, Wang YC, Jones HM, Campana D, Coustan-Smith E, Raimondi SC, Hirsch B, Hitzler JK, Head DR

Arch Pathol Lab Med. 2020 Apr;144(4):466-472. doi: 10.5858/arpa.2018-0526-OA. Epub 2019 Aug 20.

Mixed-phenotype acute leukemia: A cohort and consensus research strategy from the Children's Oncology Group Acute Leukemia of Ambiguous Lineage Task Force
Orgel E, Alexander TB, Wood BL, Kahwash SB, Devidas M, Dai Y, Alonzo TA, Mullighan CG, Inaba H, Hunger SP, Raetz EA, Gamis AS, Rabin KR, Carroll AJ 3rd, Heerema NA, Berman JN, Woods WG, Loh ML, Zweidler-McKay PA, Horan JT; Children's Oncology Group Acute Leukemia of Ambiguous Lineage Task Force

Cancer. 2020 Feb 1;126(3):593-601. doi: 10.1002/cncr.32552. Epub 2019 Oct 29

Genome Editing in Zebrafish Using High-Fidelity Cas9 Nucleases: Choosing the Right Nuclease for the Task
Prykhozhij SV, Cordeiro-Santanach A, Caceres L, Berman JN

Methods Mol Biol. 2020;2115:385-405. doi: 10.1007/978-1-0716-0290-4_21.

The Zebrafish Xenograft Platform-A Novel Tool for Modeling KSHV-Associated Diseases
Pringle ES, Wertman J, Melong N, Coombs AJ, Young AL, O'Leary D, Veinotte C, Robinson CA, Ha MN, Dellaire G, Druley TE, McCormick C, Berman JN

Viruses. 2019 Dec 20;12(1):12. doi: 10.3390/v12010012

Humanized zebrafish enhance human hematopoietic stem cell survival and promote acute myeloid leukemia clonal diversity
Rajan V, Melong N, Wong WH, King B, Tong RS, Mahajan N, Gaston D, Lund T, Rittenberg D, Dellaire G, Campbell CJV, Druley T, Berman JN

Haematologica. 2019 Dec 19:haematol.2019.223040. doi: 10.3324/haematol.2019.223040. Online ahead of print.

Frizzled 4 regulates ventral blood vessel remodeling in the zebrafish retina
Caceres L, Prykhozhij SV, Cairns E, Gjerde H, Duff NM, Collett K, Ngo M, Nasrallah GK, McMaster CR, Litvak M, Robitaille JM, Berman JN

Dev Dyn. 2019 Dec;248(12):1243-1256. doi: 10.1002/dvdy.117. Epub 2019 Oct 21

High-throughput Chemical Screening Identifies Focal Adhesion Kinase and Aurora Kinase B Inhibition as a Synergistic Treatment Combination in Ewing Sarcoma
Wang S, Hwang EE, Guha R, O'Neill AF, Melong N, Veinotte CJ, Conway Saur A, Wuerthele K, Shen M, McKnight C, Alexe G, Lemieux ME, Wang A, Hughes E, Xu X, Boxer MB, Hall MD, Kung A, Berman JN, Davis MI, Stegmaier K, Crompton BD

Clin Cancer Res. 2019 Jul 15;25(14):4552-4566. doi: 10.1158/1078-0432.CCR-17-0375. Epub 2019 Apr 12.

Fats enhance stem cell emergence.
Rajan V, Berman JN.

Science. 2019 Mar 8;363(6431):1041-1042. doi: 10.1126/science.aaw7059.

Optimized knock-in of point mutations in zebrafish using CRISPR/Cas9
Prykhozhij SV, Fuller C, Steele SL, Veinotte CJ, Razaghi B, Robitaille JM, McMaster CR, Shlien A, Malkin D, Berman JN.

Nucleic Acids Res. 2018 Sep 28;46(17):e102. doi: 10.1093/nar/gky512.PMID: 29905858 Free PMC article.

Zebrafish knock-ins swim into the mainstream
Prykhozhij SV, Berman JN.

Dis Model Mech. 2018 Oct 24;11(10):dmm037515. doi: 10.1242/dmm.037515.

Etiology and functional validation of gastrointestinal motility dysfunction in a zebrafish model of CHARGE syndrome
Cloney K, Steele SL, Stoyek MR, Croll RP, Smith FM, Prykhozhij SV, Brown MM, Midgen C, Blake K, Berman JN.

FEBS J. 2018 Jun;285(11):2125-2140. doi: 10.1111/febs.14473. Epub 2018 Apr 27.

How Surrogate and Chemical Genetics in Model Organisms Can Suggest Therapies for Human Genetic Diseases
Strynatka KA, Gurrola-Gal MC, Berman JN, McMaster CR.

Genetics. 2018 Mar;208(3):833-851. doi: 10.1534/genetics.117.300124.

hace1 Influences zebrafish cardiac development via ROS-dependent mechanisms
Razaghi B, Steele SL, Prykhozhij SV, Stoyek MR, Hill JA, Cooper MD, McDonald L, Lin W, Daugaard M, Crapoulet N, Chacko S, Lewis SM, Scott IC, Sorensen PHB, Berman JN.

Dev Dyn. 2018 Feb;247(2):289-303. doi: 10.1002/dvdy.24600. Epub 2017 Oct 27.

Loss of PRP4K drives anoikis resistance in part by dysregulation of epidermal growth factor receptor endosomal trafficking
Corkery DP, Clarke LE, Gebremeskel S, Salsman J, Pinder J, Le Page C, Meunier L, Xu Z, Mes-Masson AM, Berman JN, Johnston B, Dellaire G.

Oncogene. 2018 Jan 11;37(2):174-184. doi: 10.1038/onc.2017.318. Epub 2017 Sep 11.

Phloridzin docosahexaenoate, a novel flavonoid derivative, suppresses growth and induces apoptosis in T-cell acute lymphoblastic leukemia cells
Arumuggam N, Melong N, Too CK, Berman JN, Rupasinghe HV.

Am J Cancer Res. 2017 Dec 1;7(12):2452-2464. eCollection 2017.

In Vivo Validation of PAPSS1 (3'-phosphoadenosine 5'-phosphosulfate synthase 1) as a Cisplatin-sensitizing Therapeutic Target.
Leung AWY, Veinotte CJ, Melong N, Oh MH, Chen K, Enfield KSS, Backstrom I, Warburton C, Yapp D, Berman JN, Bally MB, Lockwood WW

Clin Cancer Res. 2017 Nov 1;23(21):6555-6566. doi: 10.1158/1078-0432.CCR-17-0700. Epub 2017 Aug 8.

Enzalutamide inhibits testosterone-induced growth of human prostate cancer xenografts in zebrafish and can induce bradycardia.
Melong N, Steele S, MacDonald M, Holly A, Collins CC, Zoubeidi A, Berman JN, Dellaire G.

Sci Rep. 2017 Oct 31;7(1):14698. doi: 10.1038/s41598-017-14413-w.

Improved outcomes for myeloid leukemia of Down syndrome: a report from the Children's Oncology Group AAML0431 trial.
Taub JW, Berman JN, Hitzler JK, Sorrell AD, Lacayo NJ, Mast K, Head D, Raimondi S, Hirsch B, Ge Y, Gerbing RB, Wang YC, Alonzo TA, Campana D, Coustan-Smith E, Mathew P, Gamis AS.

Blood. 2017 Jun 22;129(25):3304-3313. doi: 10.1182/blood-2017-01-764324. Epub 2017 Apr 7.

A rapid and effective method for screening, sequencing and reporter verification of engineered frameshift mutations in zebrafish.
Prykhozhij SV, Steele SL, Razaghi B, Berman JN.

Dis Model Mech. 2017 Jun 1;10(6):811-822. doi: 10.1242/dmm.026765. Epub 2017 Mar 9.

Using zebrafish models of leukemia to streamline drug screening and discovery.
Deveau AP, Bentley VL, Berman JN.

Exp Hematol. 2017 Jan;45:1-9. doi: 10.1016/j.exphem.2016.09.012. Epub 2016 Oct 6.

New Developments in CRISPR/Cas-based Functional Genomics and their Implications for Research Using Zebrafish
Prykhozhij SV, Caceres L, Berman JN.

Curr Gene Ther. 2017;17(4):286-300. doi: 10.2174/1566523217666171121164132.

Roles for APRIN (PDS5B) in homologous recombination and in ovarian cancer prediction
Couturier AM, Fleury H, Patenaude AM, Bentley VL, Rodrigue A, Coulombe Y, Niraj J, Pauty J, Berman JN, Dellaire G, Di Noia JM, Mes-Masson AM, Masson JY

Nucleic Acids Res. 2016 Dec 15;44(22):10879-10897. doi: 10.1093/nar/gkw921. Epub 2016 Oct 24.

Insert, remove or replace: A highly advanced genome editing system using CRISPR/Cas9
Ceasar SA, Rajan V, Prykhozhij SV, Berman JN, Ignacimuthu S.

Biochim Biophys Acta. 2016 Sep;1863(9):2333-44. doi: 10.1016/j.bbamcr.2016.06.009. Epub 2016 Jun 24.

Characterization of the Zebrafish Homolog of β-Glucosidase 2: A Target of the Drug Miglustat.​
Sultana S, Truong NY, Vieira DB, Wigger JG, Forrester AM, Veinotte CJ, Berman JN, van der Spoel AC.​

Zebrafish. 2016 Feb 24. [Epub ahead of print].

A Guide to Computational Tools and Design Strategies for Genome Editing Experiments in Zebrafish Using CRISPR/Cas9.
Prykhozhij SV, Rajan V, Berman JN.

Zebrafish. 2016 Feb;13(1):70-3.

Glycine and Folate Ameliorate Models of Congenital Sideroblastic Anemia
J Fernández-Murray JP, Prykhozhij SV, DufayN, Steele SL, Gaston D, Nasrallah GK, Coombs AJ, Fernandez CV, Berman JN, and McMaster CR

PLoS Genetics. 2016 Jan 28;12(1).

A Guide to Computational Tools and Design Strategies for Genome Editing Experiments in ZebrafishUsing CRISPR/Cas9
Prykhozhij SV, Rajan V, and Berman JN

TechnoFish-Methods. Zebrafish (in press)

Bone marrow failure and developmental delay caused by mutations in poly(A)-specific ribonuclease (PARN)
Dhanraj S, Gunja SM, Deveau AP, Nissbeck M, Boonyawat B, Coombs AJ, Renieri A, Mucciolo M, Marozza A, Buoni S, Turner L, Li H, Jarrar A, Sabanayagam M, Kirby M, Shago M, Pinto D, Berman JN, Scherer SW, Virtanen A, Dror Y

J Med Genet. 2015 Nov;52(11):738-48.

Epigenetic therapy restores normal hematopoiesis in a zebrafish model of NUP98-HOXA9-induced myeloid disease
Deveau AP, Forrester AM, Coombs AJ, Wagner GS, Grabher C, Chute I, Leger D, Mingay M, Hirst M, Lewis S, Look AT, and Berman JN

Leukemia 2015 Oct;29(10):2086-9.

Translational activation of HIF1α by YB-1 promotes sarcoma metastasis
El-Naggar AM, Veinotte CJ, Tognon CE, Corkery DP Cheng H, Tirode F, Grunewald TGP, Kyle A, Baker JH, Mathers J, Somasekharan SP, LePard NE, McKinney S, Bennewith KL, Minchinton AI, Delattre O, Wang Y, Dellaire G, Berman JN, and Sorensen PH

Cancer Cell 2015 May 11;27(5):682-97.

CRISPR MultiTargeter: a web tool to find common and unique CRISPR single guide RNA targets in a set of similar sequences
Prykhozhij SV, Rajan V, Gaston D, and Berman JN

PLOS One 2015 Mar 5;10(3):e0119372.

Visnagin protects against doxorubicin-induced cardiomyopathy through modulation of mitochondrial malate dehydrogenase
Liu Y, Asnani A, Zou L, Bentley VL, Yu M, Wang Y, Dellaire G, Sarkar KS, Dai M, Chen HH, Sosnovik DE, Shin JT, Haber DA, Berman JN, Chao W, and Peterson RT

Sci Transl Med. 2014 Dec 10;6(266):266ra170.

Zebrafish Xenotransplantation and Focused Chemical Genomics: A Preclinical Therapeutic Model For T-Cell Acute Lymphoblastic Leukemia. Haematologica
Bentley VL, Veinotte CJ, Corkery D, Pinder J, Leblanc MA, Bedard K, Weng AP, Berman JN* and Dellaire G*

2015 Jan;100(1):70-6. *Co-corresponding authors

Optimized cell transplantation using hypomorphic rag2 mutant zebrafish
Tang Q, Abdelfattahi NS, Blackburn JS, Moore JC, Martinez SA, Moore FE, Lobbardi R, Tenente IM, Ignatius MS, Berman JN, Liwksi RS, Houvras Y, and Langenau1, DM

Nat Methods. 2014 Aug;11(8):821-4. doi: 10.1038/nmeth.3031. Epub 2014 Jul 20.

Hooking the Big One: The Potential of Zebrafish Xenotransplantation to Reform Cancer Drug Screening in the Genomic Era
Veinotte CJ, Dellaire G, and Berman JN

Dis Mod Mech 2014 Jul;7(7):745-54.

A transgenic zebrafish model expressing KIT-D816V recapitulates features of aggressive systemic mastocytosis
Balci TB, Prykhozhij SV, Teh EM, Da’as SI, McBride E, Liwski R, Chute IC, Leger D, Lewis SM, and Berman JN

Br J Haematol 2014 Oct;167(1):48-61. doi: 10.1111/bjh.12999. Epub 2014 Jul 2.

The progress and promise of zebrafish as a model to study mast cells
Prykhozhij SV, Berman JN

Dev Comp Immunol 2014 Sep;46(1):74-83.

Calcineurin-Rcan1 interaction contributes to stem cell factor-mediated mast cell activation
Wu Z, Macneil AJ, Junkins R, Berman J. N, and Lin TJ

 J Immunol, 2013 Dec 15;191(12):5885-94.

Zebrafish as a model system for mitochondrial biology and diseases. Translational Research
Steele, S. L., Prykhozhij, S. V., & Berman, J. N

Translational Research. 2013 Sep 23. doi:10.1016/j.trsl.2013.08.008

Hace1 controls ROS generation of vertebrate Rac1-dependent NADPH oxidase complexes
Daugaard, M., Nitsch, R., Razaghi, B., McDonald, L., Jarrar, A., Torrino, S., Castillo-Lluva, S., Rotblat, B., Li, L., Malliri, A., Lemichez, E., Mettouchi, A., Berman, J. N., Penninger, J. M., & Sorensen, P. H

Nature Commun. 2013 Jul 17. doi:10.1038/ncomms3180.

Zebrafish xenografts as a tool for in vivo studies on human cancer
Konantz M, Balci TB, Hartwig UF, Dellaire G, André MC, Berman JN, Lengerke C

Ann N Y Acad Sci. 2012 Aug;1266:124-37. doi: 10.1111/j.1749-6632.2012.06575.x. Review.

The zebrafish reveals dependence of the mast cell lineage on Notch signaling in vivo
Da’as SI, Coombs AJ, Balci TB, Grondin CA, Ferrando AA, Berman JN

Blood. 2012 Apr 12;119(15):3585-94. doi: 10.1182/blood-2011-10-385989. Epub 2012 Feb 24.

A unique role of GATA1s in Down syndrome acute megakaryocytic leukemia biology and therapy
Xavier AC, Edwards H, Dombkowski AA, Balci TB, Berman JN, Dellaire G, Xie C, Buck SA, Matherly LH, Ge Y, Taub JW

PloS one. 2011 Nov 6.11: e27486. doi: 10.1371/journal.pone.0027486

NUP98-HOXA9-transgenic zebrafish develop a myeloproliferative neoplasm and provide new insight into mechanisms of myeloid leukaemogenesis
Forrester AM, Grabher C, McBride ER, Boyd ER, Vigerstad MH, Edgar A, Kai FB, Da’as SI, Payne E, Look AT, Berman JN

Br J Haematol. 2011 Aug 2. doi: 10.1111/j.1365-2141.2011.08810.x.

Using MS-MLPA as an efficient screening tool for detecting 9p21 abnormalities in pediatric acute lymphoblastic leukemia
Gardiner RB, Morash BA, Riddell C, Wang H, Fernandez CV, Yhap M, Berman JN

Pediatr Blood Cancer. 2011 Jul 27. doi: 10.1002/pbc.23285.

Regulator of calcineurin 1 (rcan1) is required for the development of pulmonary eosinophilia in allergic inflammation in mice
Yang YJ, Macneil AJ, Junkins R, Carrigan SO, Tang JT, Forward N, Hoskin D, Berman JN, Lin TJ

Am J Pathol. 2011 Jul 8.

Leukaemia xenotransplantation in zebrafish – chemotherapy response assay in vivo
Corkery DP, Dellaire G, Berman JN

Br J Haematol. 2011 Jun;153(6):786-9. doi: 10.1111/j.1365-2141.2011.08661.x. Epub 2011 Apr 22.

Zebrafish mast cells demonstrate conserved innate and adaptive immune responses
Da’as S, Teh EM, Dobson JT, Nasrallah GK, McBride ER, Wang H, Neuberg DS, Marshall JS, Lin TJ, Berman JN

Dev Comp Immunol. 2011 Jan; 35(1):125-34

The Hox cofactors Meis1 and Pbx act upstream of gata1 to regulate primitive hematopoiesis
Pillay LM, Forrester AM, Erickson T, Berman JN, Waskiewicz AJ

Dev Biol. 2010 Apr 15;340(2):306-17.

Cladribine inhibits a diltiazem-induced increase in red blood cell purine nucleotide concentrations in a zebrafish model
Klein LC, Yeung PK, Berman JN

Biomarkers. 2009 Dec;14(8):554-9

The impact of prophylactic fresh-frozen plasma and cryoprecipitate on the incidence of central nervous system thrombosis and hemorrhage in children with acute lymphoblastic leukemia receiving asparaginase
Abbott LS, Deevska M, Fernandez CV, Dix D, Price VE, Wang H, Parker L, Yhap M, Fitzgerald C, Barnard DR, Berman JN

Blood. 2009 Dec 10;114(25):5146-51.

Fluorescence-activated cell sorting (FACS) of whole mount in situ hybridization (WISH) labelled haematopoietic cell populations in the zebrafish
Dobson JT, Da’as S, McBride ER, Berman JN.

 Br J Haematol. 2009 Mar;144(5):732-5.

Rcan1 negatively regulates Fc epsilonRI-mediated signaling and mast cell function
Yang YJ, Chen W, Edgar A, Li B, Molkentin JD, Berman JN, Lin TJ

J Exp Med. 2009 Jan 16;206(1):195-207. Epub 2009 Jan 5. PubMed PMID: 19124655; PubMed Central PMCID: PMC2626669.

TRAF6 specifically contributes to FcepsilonRI-mediated cytokine production but not mast cell degranulation
Yang YJ, Chen W, Carrigan SO, Chen WM, Roth K, Akiyama T, Inoue J, Marshall JS, Berman JN, Lin TJ

J Biol Chem. 2008 Nov 14;283(46):32110-8. Epub 2008 Sep 4.

Carboxypeptidase A5 identifies a novel mast cell lineage in the zebrafish providing new insight into mast cell fate determination
Dobson JT, Seibert J, Teh EM, Da’as S, Fraser RB, Paw BH, Lin TJ, Berman JN

Blood. 2008 Oct 1;112(7):2969-72. Epub 2008 Jul 17.

JAK2 V617F positive polycythemia Vera in a child with neurofibromatosis type I
Berman JN, Greer WL, Archambeault S, Loh ML, Riddell C, Morash B, Dumas N, Fernandez CV, Ludman MD

Pediatr Blood Cancer. 2008 Nov;51(5):689-91.

Routine bone marrow examination in the initial evaluation of paediatric Hodgkin lymphoma: the Canadian perspective
Simpson CD, Gao J, Fernandez CV, Yhap M, Price VE, Berman JN

Br J Haematol. 2008 Jun;141(6):820-6.

Targeting transcription factors in acute leukemia in children
Berman JN, Look AT

Curr Drug Targets. 2007 Jun;8(6):727-37. Review.

Current and future approaches for the therapeutic targeting of metastasis (review)
Germanov E, Berman JN, Guernsey DL

Int J Mol Med. 2006 Dec;18(6):1025-36. Review.

Herpes zoster infection in the post-hematopoietic stem cell transplant pediatric population may be preceded by transaminitis: an institutional experience
Berman JN, Wang M, Berry W, Neuberg DS, Guinan EC

 Bone Marrow Transplant. 2006 Jan;37(1):73-80.

Zebrafish as a model for myelopoiesis during embryogenesis
Berman JN, Kanki JP, Look AT

Exp Hematol. 2005 Sep;33(9):997-1006. Review.

CD34+ cell selection is required to assess HOXA9 expression levels in patients with myelodysplastic syndrome
Heinrichs S, Berman JN, Ortiz TM, Kornblau SM, Neuberg DS, Estey EH, Look AT

Br J Haematol. 2005 Jul;130(1):83-6.

CD34+ cell selection is required to assess HOXA9 expression levels in patients with myelodysplastic syndrome
The pu.1 promoter drives myeloid gene expression in zebrafish

Blood. 2004 Sep 1;104(5):1291-7. Epub 2004 Mar 2.

MacLean JE, Wertman JN, Prykhozhij SV, Chedrawe E, Langley S, Steele SL, Ban K, Blake K, Berman JN.

Genes (Basel). 2023 May 15;14(5):1086. doi: 10.3390/genes14051086.

Genetic Models of Rare Diseases

 

Zebrafish are a remarkably versatile platform for studying rare diseases (RD). The Berman lab studies genetic lesions that cause limb-girdle muscular dystrophy (LGMD). LGMD are a group of RDs that cause weakness and wasting of the muscles most proximal to the trunk, with multiple subtypes and different inheritance patterns that include mutations in LMNA, CAPN3, and DYSF genes. To assess zebrafish phenotypes, we use the birefringence assay and functional assays depending on the disease being modeled. Zebrafish models of LGMD and other muscular dystrophies have been generated to perform drug screens with chemical libraries to identify promising therapies.

The Berman lab also studies familial exudative vitreoretinopathy (FEVR), a rare congenital disorder commonly caused by mutations of the FZD4 gene and other genes coding for proteins in the Norrin signaling pathway. FEVR is characterized by a lack of blood vessel growth to the periphery of the retina causing leakage and hemorrhage and resulting in retinal detachment and blindness. We have generated a visually impaired zebrafish FEVR model exhibiting abnormal retinal vasculature to study vascular biology in retinovascular disorders and improve patient outcomes.

Lastly, CHARGE syndrome is linked to autosomal-dominant mutations in the CHD7 gene and results in multiple physiological and structural abnormalities, including heart defects, hearing and vision loss, and gastrointestinal (GI) problems. Our zebrafish CHARGE models have allowed us to examine GI structure, innervation, and motility, along with heart rate and responses to anesthesia. These zebrafish models help us to better understand the biological and genetic causes of pathology in these diseases and provide a platform for high-throughput drug screening.

Xenotransplantation Models of Pediatric Cancers​

 

The Berman lab has pioneered the transplantation of human cancer cells into the larval zebrafish for novel drug discovery and improving current clinical chemotherapies. We transplant both human cancer cell lines and patient derived tumour cells into larval zebrafish to better understand cancer progression and to find more personalized therapies. Cells can be tracked in real-time for proliferation, migration and homing to tissue niches. Responses to therapeutics added to the larval water or administered by intraperitoneal injection or gavage can be evaluated by fluorescence microscopy and quantified using ex vivo cell enumeration.

This platform has already been adapted to study metastasis in sarcoma; test novel compounds in AML; and reveal drug response-tumour genotype correlations in primary patient-derived T-cell acute lymphoblastic leukemia (ALL). This model has the potential to provide relevant drug response data in a clinically actionable timeframe, which can help improve patient outcomes and aid in a better quality of life. The Berman lab has leveraged this platform in the Model Systems Node of the Terry Fox Research Institute PRecision Oncology For Young peopLE (PROFYLE) program, which is an innovative pan-Canadian medicine program that strives to create new therapeutic inroads for children and youth with refractory, relapsed, or metastatic childhood cancers. The PROFYLE program aims to molecularly characterize 450 hard-to-treat cancers from children across Canada, experimentally model these tumours, and find molecularly targeted and biologically active treatments.

Genetic Models of Cancer and Cancer Predisposition Syndromes​

 

The Berman lab uses transgenic and CRISPR based approaches in the zebrafish to model cancer mutations and cancer predisposition syndromes to better understand the molecular mechanisms underpinning childhood leukemia and other cancers. We have developed and study zebrafish models of Li-Fraumeni Syndrome (LFS), a devastating cancer predisposition syndrome most frequently caused by autosomal inheritance of germline mutations in the TP53 gene.

We also study and have generated models of inherited bone marrow failure syndrome (IBMFS), which are characterized by deficient hematopoiesis in a subset of blood lineages and predispose children to developing myelodysplastic syndrome (MDS) and acute myeloid leukemia (AML). Shwachman-Diamond syndrome (SDS) is a well-known IBMFS, commonly caused by germline mutations in the SBDS gene and less frequently in the DNAJC21 gene. Affected children have an increased risk of developing MDS and AML.

We have been developing a model of DNAJC21-deficient SDS. Infant leukemia (IL) is a rare and aggressive leukemia that progresses rapidly with significant treatment and management challenges. Reciprocal translocations of the MLL1 (KMT2A) gene are present in most cases of either infant AML or infant acute lymphoblastic leukemia (ALL). We have generated the first IL zebrafish model by expressing MLL1 gene fusions in pathologically relevant blood cells. Our goal for these models is to increase the biological understanding of these diseases and to perform high-throughput phenotype-based in vivo testing of small molecules and targeted therapies that ameliorate disease and would otherwise be infeasible in other model systems.