- Developmental Biology and Gene Regulation
- Hedgehog Signaling Pathway Studies
- Congenital heart defects research
- Congenital Ear and Nasal Anomalies
- Genomic variations and chromosomal abnormalities
- Epigenetics and DNA Methylation
- Zebrafish Biomedical Research Applications
- Craniofacial Disorders and Treatments
- Wnt/β-catenin signaling in development and cancer
- Genetics and Neurodevelopmental Disorders
- Cancer-related Molecular Pathways
- melanin and skin pigmentation
- RNA regulation and disease
- Neuroscience and Neural Engineering
- Silk-based biomaterials and applications
- Ion channel regulation and function
- Autism Spectrum Disorder Research
- Cell Adhesion Molecules Research
- Pluripotent Stem Cells Research
- RNA Research and Splicing
- Neural dynamics and brain function
- Protist diversity and phylogeny
- Connective tissue disorders research
- Linguistic, Cultural, and Literary Studies
- Muscle Physiology and Disorders
University of Wisconsin–Madison
2013-2024
Smith-Kettlewell Eye Research Institute
2017-2019
Kanazawa University
2009
Whitehead Institute for Biomedical Research
1996-2004
Massachusetts Institute of Technology
1998-2001
Harvard University
1988-1994
The promoter from the metallothiortein gene may be a useful conditional for construction of chimeric genes to expressed in Drosophila cells culture. To explore this possibility responses endogenous metallothionein and an vitro constructed containing were examined. Copper cadmium, when added growth medium Schneider's line 2 cells, can produce 30–100 fold induction mRNA levels. level depends on amount copper or cadmium these levels remain high at least four days. is less toxic than does not...
Wnt and Hedgehog signaling pathways play central roles in embryogenesis, stem cell maintenance, tumorigenesis. However, the mechanisms by which these two interact are not well understood. Here, we identified a novel mechanism pathway stimulates transcriptional output of signaling. Wnt/beta-catenin induces expression an RNA-binding protein, CRD-BP, turn binds stabilizes GLI1 mRNA, causing elevation activity. The newly described mode regulation seems to be important several functions Wnt,...
ABSTRACT We report an analysis of forebrain determination and patterning in the zebrafish Danio rerio. In order to study these events, we isolated homologs two neural markers, odd-paired-like (opl), which encodes a zinc finger protein, fkh5, forkhead domain protein. At mid-gastrula, expression genes defines very early pattern presumptive neurectoderm, with opl later expressed telencephalon, fkh5 diencephalon more posterior neurectoderm. Using vitro explant assays, show that induction has...
ABSTRACT We report the first extended culture system for analysing zebrafish (Danio rerio) embryogenesis with which we demonstrate neural induction and anteroposterior patterning. Explants from animal pole region of blastula embryos (‘animal caps’) survived at least two days increased in cell number. Mesodermal neural-specific genes were not expressed cultured caps, although low levels dorsoanterior marker otx2 seen. In contrast, observed strong expression gta3, a ventral cyt1, novel type I...
Alternative cleavage and polyadenylation generates multiple transcript variants producing mRNA isoforms with different length 3′-UTRs. enables differential post-transcriptional regulation via the availability of cis-acting elements in Microphthalmia-associated transcription factor (MITF) is a master regulator melanocyte development melanogenesis. This central also implicated melanoma development. Here, we show that cells favor expression MITF shorter 3′-UTR. We establish this isoform...
Wnt growth factors acting through the canonical intracellular signaling cascade play fundamental roles during vertebrate brain development. In particular, is crucial for normal development of dorsal midbrain, future optic tectum. Wnts act both as patterning signals and regulators cell growth. developing tectum, mitogenic; however, mechanism function not known. As a step towards better understanding this mechanism, we have identified two new targets, closely linked zic2a zic5 genes. Using...
Anteroposterior (A/P) patterning of the vertebrate neurectoderm begins early during development. In vitro explant assays have been used to show that an A/P subdivision zebrafish is in place by gastrulation. However, no direct markers this had described. We report isolation two members zic gene family, zic2 and zic3, which mark distinct regions developing nervous system. zic3 expressed gastrula embryos a posterior domain, directly demonstrating pattern present future stage. Analysis mutants...
ABSTRACT The Drosophila position-specific (PS) antigens are homologues of the vertebrate integrins, a family trans-membrane proteins that function in cell-matrix and cell-cell adhesion. common β subunit PS integrins (PSβ) is encoded by lethal(l)myospheroid gene (mys) required during wing, eye muscle development. By expressing PSβ protein at defined developmental periods, we have shown throughout pupation, but not earlier, for normal development wings. In contrast, key requirement occurs only...
The ability to filter sensory information into relevant versus irrelevant stimuli is a fundamental, conserved property of the central nervous system and accomplished in part through habituation learning. Synaptic plasticity that underlies learning has been described at cellular level, yet genetic regulators this remain poorly understood, as do circuits mediate filtering.To identify genes critical for plasticity, forward screen zebrafish was performed, which identified mutant allele,...
Holoprosencephaly (HPE) is the most common congenital malformation of forebrain in human. Several genes with essential roles during development have been identified because they cause HPE when mutated. Among these are that encode secreted growth factor Sonic hedgehog (Shh)and transcription factors Six3 and Zic2. In mouse, Shh activate each other's transcription, but a role for Zic2 this interaction has not tested. We demonstrate zebrafish, as Hh signaling activates six3b developing...
During neurulation, vertebrate embryos form a neural tube (NT), the rudiment of central nervous system. In mammals and birds, key step in cranial NT morphogenesis is dorsolateral hinge-point (DLHP) bending, which requires an apical actomyosin network. The mechanism DLHP formation poorly understood, although several essential genes have been identified, among them Zic2, encodes zinc-finger transcription factor. We found that zebrafish midbrain also Zic function. Given this conservation, we...
Abstract Members of the Wnt family extracellular proteins play essential roles during many phases vertebrate embryonic development. The molecular mechanism their action involves a complex cascade intracellular signaling events, which remains to be understood completely. Recently, two novel cytoplasmic modulators signaling, Frodo and Dapper, were identified in Xenopus . We report isolation homologs zebrafish, show that these genes, frd1 frd2 , are expressed restricted domains embryogenesis....
Journal Article Isolation and characterization of the Drosophila translational elongation factor 2 gene Get access Yevgenya Grinblat, Grinblat 1The Biological Laboratones, Harvard University16 Divinity Avenue, Cambridge, MA 02138, USA Search for other works by this author on: Oxford Academic PubMed Google Scholar Nicholas H. Brown, Brown Fotis C. Kafatos USA2Institute Molecular Biology Biotechnology, Research Center Crete Department Biology, University CreteHeraklion, 711 10 Crete, Greece...
Factor for adipocyte differentiation 24 (fad24) is a novel gene that has been implicated in and DNA replication. In screen zebrafish mutants have an abnormal tissue distribution of neutrophils, we identified insertional allele fad24, fad24hi1019. Homozygous fad24hi1019 larvae exhibit muscle degeneration accompanied by leukocyte infiltration. Muscle was extensive included apoptosis disorganized, poorly striated fibers. Blocking using pan-caspase inhibitors resulted decreased neutrophil...
Article11 September 2020Open Access Source DataTransparent process ALX1-related frontonasal dysplasia results from defective neural crest cell development and migration Jonathan Pini Center for Regenerative Medicine, Department of Surgery, Massachusetts General Hospital, Boston, MA, USA Shriners Hospital Children, Search more papers by this author Janina Kueper Institute Human Genetics, University Bonn, Germany Correction added on 7 July 2022, after first online publication: The affiliation...
Abstract To identify genes required for development of the brain and somites, we performed a pilot screen gynogenetic haploid zebrafish embryos produced from mothers mutagenized by viral insertion. We describe an efficient method to new mutations affected gene. In addition, report results small‐scale that identified five development, including novel alleles nagie oko, pou5f1, ribosomal protein L36 , n‐cadherin as well allele laminin g1 gene is normal skeletal muscle fiber organization somite...
Cilia are cell surface organelles with key roles in a range of cellular processes, including generation fluid flow by motile cilia. The axonemes cilia and immotile kinocilia contain 9 peripheral microtubule doublets, central pair, connecting radial spokes. Aberrant spoke components RSPH1, 3, 4a have been linked primary ciliary dyskinesia (PCD), disorder characterized dysmotility; yet, functions remain unclear. Here we show that zebrafish Rsph9 is expressed cells bearing kinocilia, localizes...
Background: Rfx winged‐helix transcription factors, best known as key regulators of core ciliogenesis, also play ciliogenesis‐independent roles during neural development. Mammalian Rfx4 controls tube morphogenesis via both mechanisms. Results: We set out to identify conserved aspects rfx 4 gene function vertebrate development and establish a new genetic model in which analyze these mechanisms further. To this end, we have generated frame‐shift alleles the zebrafish locus using CRISPR/Cas9...
Rett syndrome (RTT), a human neurodevelopmental disorder characterized by severe cognitive and motor impairments, is caused dysfunction of the conserved transcriptional regulator Methyl-CpG-binding protein 2 (MECP2). Genetic analyses in mouse Mecp2 mutants, which exhibit key features RTT, have been essential for deciphering mechanisms MeCP2 function; nonetheless, our understanding these complex incomplete. Zebrafish mecp2 mutants mild behavioral deficits but not analyzed depth. Here, we...