Program in Developmental Biology,
Department of Molecular and Human Genetics,
Department of Neuroscience
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Sequoia regulates cell fate decisions in the external sensory organs of adult Drosophila.
EMBO reports Jun, 2009 | Pubmed ID: 19444309
Endocytosis and intracellular trafficking of Notch and its ligands.
Current topics in developmental biology , 2010 | Pubmed ID: 20816395
dEHBP1 controls exocytosis and recycling of Delta during asymmetric divisions.
The Journal of cell biology Jan, 2012 | Pubmed ID: 22213802
A mutation in EGF repeat-8 of Notch discriminates between Serrate/Jagged and Delta family ligands.
Science (New York, N.Y.) Nov, 2012 | Pubmed ID: 23197537
Crag is a GEF for Rab11 required for rhodopsin trafficking and maintenance of adult photoreceptor cells.
PLoS biology , 2012 | Pubmed ID: 23226104
The C8ORF38 homologue Sicily is a cytosolic chaperone for a mitochondrial complex I subunit.
The Journal of cell biology Mar, 2013 | Pubmed ID: 23509070
Drosophila EHBP1 regulates Scabrous secretion during Notch-mediated lateral inhibition.
Journal of cell science Aug, 2013 | Pubmed ID: 23788431
Protein phosphatase 1ß limits ring canal constriction during Drosophila germline cyst formation.
PloS one , 2013 | Pubmed ID: 23936219
Drosophila Tempura, a novel protein prenyltransferase α subunit, regulates notch signaling via Rab1 and Rab11.
PLoS biology Jan, 2014 | Pubmed ID: 24492843
Shared mechanisms between Drosophila peripheral nervous system development and human neurodegenerative diseases.
Current opinion in neurobiology Aug, 2014 | Pubmed ID: 24762652
Dopamine dynamics and signaling in Drosophila: an overview of genes, drugs and behavioral paradigms.
Experimental animals , 2014 | Pubmed ID: 24770636
The retromer complex is required for rhodopsin recycling and its loss leads to photoreceptor degeneration.
PLoS biology Apr, 2014 | Pubmed ID: 24781186
Introduction to Notch signaling.
Methods in molecular biology (Clifton, N.J.) , 2014 | Pubmed ID: 25053477
Large-scale identification of chemically induced mutations in Drosophila melanogaster.
Genome research Oct, 2014 | Pubmed ID: 25258387
A drosophila genetic resource of mutants to study mechanisms underlying human genetic diseases.
Cell Sep, 2014 | Pubmed ID: 25259927
Mitochondrial fusion but not fission regulates larval growth and synaptic development through steroid hormone production.
eLife Oct, 2014 | Pubmed ID: 25313867
Glial lipid droplets and ROS induced by mitochondrial defects promote neurodegeneration.
Cell Jan, 2015 | Pubmed ID: 25594180
Fruit flies in biomedical research.
Genetics Mar, 2015 | Pubmed ID: 25624315
A voltage-gated calcium channel regulates lysosomal fusion with endosomes and autophagosomes and is required for neuronal homeostasis.
PLoS biology Mar, 2015 | Pubmed ID: 25811491
Impaired Mitochondrial Energy Production Causes Light-Induced Photoreceptor Degeneration Independent of Oxidative Stress.
PLoS biology Jul, 2015 | Pubmed ID: 26176594
Morgan's legacy: fruit flies and the functional annotation of conserved genes.
Cell Sep, 2015 | Pubmed ID: 26406362
WAC Regulates mTOR Activity by Acting as an Adaptor for the TTT and Pontin/Reptin Complexes.
Developmental cell Jan, 2016 | Pubmed ID: 26812014
Ubr3, a Novel Modulator of Hh Signaling Affects the Degradation of Costal-2 and Kif7 through Poly-ubiquitination.
PLoS genetics 05, 2016 | Pubmed ID: 27195754
Loss of Frataxin induces iron toxicity, sphingolipid synthesis, and Pdk1/Mef2 activation, leading to neurodegeneration.
eLife 06, 2016 | Pubmed ID: 27343351
Rare Functional Variant in TM2D3 is Associated with Late-Onset Alzheimer's Disease.
PLoS genetics Oct, 2016 | Pubmed ID: 27764101
A Syndromic Neurodevelopmental Disorder Caused by De Novo Variants in EBF3.
American journal of human genetics Jan, 2017 | Pubmed ID: 28017372
Loss of Nardilysin, a Mitochondrial Co-chaperone for α-Ketoglutarate Dehydrogenase, Promotes mTORC1 Activation and Neurodegeneration.
Neuron Jan, 2017 | Pubmed ID: 28017472
MARRVEL: Integration of Human and Model Organism Genetic Resources to Facilitate Functional Annotation of the Human Genome.
American journal of human genetics Jun, 2017 | Pubmed ID: 28502612
Neutral Competition for Drosophila Follicle and Cyst Stem Cell Niches Requires Vesicle Trafficking Genes.
Genetics Jul, 2017 | Pubmed ID: 28512187
Clinically severe CACNA1A alleles affect synaptic function and neurodegeneration differentially.
PLoS genetics Jul, 2017 | Pubmed ID: 28742085
Model Organisms Facilitate Rare Disease Diagnosis and Therapeutic Research.
Genetics 09, 2017 | Pubmed ID: 28874452
Pleiotropic neuropathological and biochemical alterations associated with Myo5a mutation in a rat Model.
Brain research 01, 2018 | Pubmed ID: 29217155
Biallelic Mutations in ATP5F1D, which Encodes a Subunit of ATP Synthase, Cause a Metabolic Disorder.
American journal of human genetics Mar, 2018 | Pubmed ID: 29478781
A gene-specific library for .
eLife 03, 2018 | Pubmed ID: 29565247
Functional variants in TBX2 are associated with a syndromic cardiovascular and skeletal developmental disorder.
Human molecular genetics 07, 2018 | Pubmed ID: 29726930
Integration of Drosophila and Human Genetics to Understand Notch Signaling Related Diseases.
Advances in experimental medicine and biology , 2018 | Pubmed ID: 30030826
IRF2BPL Is Associated with Neurological Phenotypes.
American journal of human genetics 08, 2018 | Pubmed ID: 30057031
Rapid and Integrative Discovery of Retina Regulatory Molecules.
Cell reports Aug, 2018 | Pubmed ID: 30157441
Effect of Genetic Diagnosis on Patients with Previously Undiagnosed Disease.
The New England journal of medicine 11, 2018 | Pubmed ID: 30304647
Unraveling Novel Mechanisms of Neurodegeneration Through a Large-Scale Forward Genetic Screen in .
Frontiers in genetics , 2018 | Pubmed ID: 30693015
Unweaving the role of nuclear Lamins in neural circuit integrity.
Cell stress Sep, 2018 | Pubmed ID: 31223139
The fruit fly at the interface of diagnosis and pathogenic mechanisms of rare and common human diseases.
Human molecular genetics Jun, 2019 | Pubmed ID: 31227826
De Novo Variants in WDR37 Are Associated with Epilepsy, Colobomas, Dysmorphism, Developmental Delay, Intellectual Disability, and Cerebellar Hypoplasia.
American journal of human genetics Aug, 2019 | Pubmed ID: 31327508
1Program in Developmental Biology, Baylor College of Medicine,
2Medical Scientist Training Program, Baylor College of Medicine,
3Department of Pediatrics, Baylor College of Medicine,
4Jan and Dan Duncan Neurological Research Institute, Texas Children's Hospital,
5Department of Molecular and Human Genetics, Baylor College of Medicine,
6Department of Neuroscience, Baylor College of Medicine,
7Howard Hughes Medical Institute, Baylor College of Medicine
J. Michael Harnish*,1,
Samantha L. Deal*,2,
Hsiao-Tuan Chao1,3,4,5,
Michael F. Wangler1,2,4,
Shinya Yamamoto1,2,4,5
1Department of Molecular and Human Genetics, Baylor College of Medicine,
2Program in Developmental Biology, Baylor College of Medicine,
3Department of Pediatrics, Section of Neurology and Developmental Neuroscience, Baylor College of Medicine,
4Jan and Dan Duncan Neurological Research Institute, Texas Children's Hospital,
5Department of Neuroscience, Baylor College of Medicine
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