Using Zebrafish to Test the Genetic Basis of Human Craniofacial Diseases

SMCHD1 mutations associated with a rare muscular dystrophy can also cause isolated arhinia and Bosma arhinia microphthalmia syndrome

(2017) Natalie D Shaw et al.   NATURE GENETICS

The old and new face of craniofacial research: How animal models inform human craniofacial genetic and clinical data

(2016) Eric Van Otterloo et al.   DEVELOPMENTAL BIOLOGY

The new NHGRI-EBI Catalog of published genome-wide association studies (GWAS Catalog)

(2016) Jacqueline MacArthur et al.   NUCLEIC ACIDS RESEARCH

Reverse Genetics in Zebrafish: Mutants, Morphants, and Moving Forward

(2016) Nathan D. Lawson   TRENDS IN CELL BIOLOGY

CRISPR Screens to Discover Functional Noncoding Elements

(2016) Jason B. Wright et al.   TRENDS IN GENETICS

Zebrafish Genome Engineering Using the CRISPR–Cas9 System

(2016) Mingyu Li et al.   TRENDS IN GENETICS

Identification of Functional Variants for Cleft Lip with or without Cleft Palate in or near PAX7, FGFR2, and NOG by Targeted Sequencing of GWAS Loci

(2015) Elizabeth J. Leslie et al.   AMERICAN JOURNAL OF HUMAN GENETICS

EFTUD2deficiency in vertebrates: Identification of a novel human mutation and generation of a zebrafish model

(2015) Brett Deml et al.   BIRTH DEFECTS RESEARCH PART A-CLINICAL AND MOLECULAR TERATOLOGY

Reverse Genetic Screening Reveals Poor Correlation between Morpholino-Induced and Mutant Phenotypes in Zebrafish

(2015) Fatma O. Kok et al.   DEVELOPMENTAL CELL

Multiplex Conditional Mutagenesis Using Transgenic Expression of Cas9 and sgRNAs

(2015) Linlin Yin et al.   GENETICS

High-throughput gene targeting and phenotyping in zebrafish using CRISPR/Cas9

(2015) Gaurav K. Varshney et al.   GENOME RESEARCH

Genetic compensation induced by deleterious mutations but not gene knockdowns

(2015) Andrea Rossi et al.   NATURE

Rapid reverse genetic screening using CRISPR in zebrafish

(2015) Arish N Shah et al.   NATURE METHODS

Functional Assessment of Disease-Associated Regulatory Variants In Vivo Using a Versatile Dual Colour Transgenesis Strategy in Zebrafish

(2015) Shipra Bhatia et al.   PLoS Genetics

EFTUD2deficiency in vertebrates: Identification of a novel human mutation and generation of a zebrafish model

(2015) Brett Deml et al.   BIRTH DEFECTS RESEARCH PART A-CLINICAL AND MOLECULAR TERATOLOGY

Multiplex Conditional Mutagenesis Using Transgenic Expression of Cas9 and sgRNAs

(2015) Linlin Yin et al.   GENETICS

Multiple genome modifications by the CRISPR/Cas9 system in zebrafish

(2014) Satoshi Ota et al.   GENES TO CELLS

Disruption of long-range gene regulation in human genetic disease: a kaleidoscope of general principles, diverse mechanisms and unique phenotypic consequences

(2014) Shipra Bhatia et al.   HUMAN GENETICS

Whole-genome duplication in teleost fishes and its evolutionary consequences

(2014) Stella M. K. Glasauer et al.   MOLECULAR GENETICS AND GENOMICS

The Serine-Threonine Protein Kinase PAK4 Is Dispensable in Zebrafish: Identification of a Morpholino-Generated Pseudophenotype

(2014) Sheran H. W. Law et al.   PLoS One

A Loss of Function Screen of Identified Genome-Wide Association Study Loci Reveals New Genes Controlling Hematopoiesis

(2014) Ewa Bielczyk-Maczyńska et al.   PLoS Genetics

Fat-Dachsous Signaling Coordinates Cartilage Differentiation and Polarity during Craniofacial Development

(2014) Pierre Le Pabic et al.   PLoS Genetics

Beyond GWASs: Illuminating the Dark Road from Association to Function

(2013) Stacey L. Edwards et al.   AMERICAN JOURNAL OF HUMAN GENETICS

FishFace: interactive atlas of zebrafish craniofacial development at cellular resolution

(2013) B Eames et al.   BMC DEVELOPMENTAL BIOLOGY

Efficient CRISPR/Cas9 genome editing with low off-target effects in zebrafish

(2013) A. Hruscha et al.   DEVELOPMENT

Functional validation of GWAS gene candidates for abnormal liver function during zebrafish liver development

(2013) L. Y. Liu et al.   Disease Models & Mechanisms

The promise of whole-exome sequencing in medical genetics

(2013) Bahareh Rabbani et al.   JOURNAL OF HUMAN GENETICS

Generation and application of signaling pathway reporter lines in zebrafish

(2013) Enrico Moro et al.   MOLECULAR GENETICS AND GENOMICS

Establishment of a bone-specific col10a1:GFP transgenic zebrafish

(2013) Yong-Il Kim et al.   MOLECULES AND CELLS

The zebrafish reference genome sequence and its relationship to the human genome

(2013) Kerstin Howe et al.   NATURE

Efficient genome editing in zebrafish using a CRISPR-Cas system

(2013) Woong Y Hwang et al.   NATURE BIOTECHNOLOGY

Genome engineering using the CRISPR-Cas9 system

(2013) F Ann Ran et al.   Nature Protocols

EMAGE mouse embryo spatial gene expression database: 2014 update

(2013) Lorna Richardson et al.   NUCLEIC ACIDS RESEARCH

Fine Tuning of Craniofacial Morphology by Distant-Acting Enhancers

(2013) C. Attanasio et al.   SCIENCE

ZFN, TALEN, and CRISPR/Cas-based methods for genome engineering

(2013) Thomas Gaj et al.   TRENDS IN BIOTECHNOLOGY

Lost in Transgenesis

(2012) Jennifer Davis et al.   CIRCULATION RESEARCH

New perspectives on pharyngeal dorsoventral patterning in development and evolution of the vertebrate jaw

(2012) Daniel Meulemans Medeiros et al.   DEVELOPMENTAL BIOLOGY

PDGFRa mutations in humans with isolated cleft palate

(2012) Sawitree Rattanasopha et al.   EUROPEAN JOURNAL OF HUMAN GENETICS

Evolution of the mammalian middle ear and jaw: adaptations and novel structures

(2012) Neal Anthwal et al.   JOURNAL OF ANATOMY

In vivo genome editing using a high-efficiency TALEN system

(2012) Victoria M. Bedell et al.   NATURE

ZFIN, the Zebrafish Model Organism Database: increased support for mutants and transgenics

(2012) Douglas G. Howe et al.   NUCLEIC ACIDS RESEARCH

Highly efficient generation of heritable zebrafish gene mutations using homo- and heterodimeric TALENs

(2012) Lindsay Cade et al.   NUCLEIC ACIDS RESEARCH

FAF1, a Gene that Is Disrupted in Cleft Palate and Has Conserved Function in Zebrafish

(2011) Michella Ghassibe-Sabbagh et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Identification of an evolutionarily conserved regulatory element of the zebrafish col2a1a gene

(2011) Rodney M. Dale et al.   DEVELOPMENTAL BIOLOGY

Bone Regenerates via Dedifferentiation of Osteoblasts in the Zebrafish Fin

(2011) Franziska Knopf et al.   DEVELOPMENTAL CELL

Examination of a palatogenic gene program in zebrafish

(2011) Mary E. Swartz et al.   DEVELOPMENTAL DYNAMICS

Genome-wide identification of conserved regulatory function in diverged sequences

(2011) L. Taher et al.   GENOME RESEARCH

Mutations in fam20b and xylt1 Reveal That Cartilage Matrix Controls Timing of Endochondral Ossification by Inhibiting Chondrocyte Maturation

(2011) B. Frank Eames et al.   PLoS Genetics

Zebrafish sp7:EGFP: A transgenic for studying otic vesicle formation, skeletogenesis, and bone regeneration

(2010) April DeLaurier et al.   GENESIS

Mutations in FAM20C also identified in non-lethal osteosclerotic bone dysplasia

(2009) MA Simpson et al.   CLINICAL GENETICS

Validating, augmenting and refining genome-wide association signals

(2009) John P. A. Ioannidis et al.   NATURE REVIEWS GENETICS

Life After GWA Studies

(2009) E. T. Dermitzakis et al.   SCIENCE

Controlling morpholino experiments: don't stop making antisense

(2008) J. S. Eisen et al.   DEVELOPMENT

Evaluation of coverage variation of SNP chips for genome-wide association studies

(2008) Mingyao Li et al.   EUROPEAN JOURNAL OF HUMAN GENETICS

Heritable targeted gene disruption in zebrafish using designed zinc-finger nucleases

(2008) Yannick Doyon et al.   NATURE BIOTECHNOLOGY

Targeted gene inactivation in zebrafish using engineered zinc-finger nucleases

(2008) Xiangdong Meng et al.   NATURE BIOTECHNOLOGY

MicroRNA Mirn140 modulates Pdgf signaling during palatogenesis

(2008) Johann K Eberhart et al.   NATURE GENETICS

Zebrafish eda and edar Mutants Reveal Conserved and Ancestral Roles of Ectodysplasin Signaling in Vertebrates

(2008) Matthew P. Harris et al.   PLoS Genetics