ArticleNeurobiology of disease2016
Postnatal and adult consequences of loss of huntingtin during development: Implications for Huntington's disease.
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Article in Neurobiology of disease, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 51 papers.
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51citing papers in PubMed
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51 citing papers in PubMed, 75 citations in OpenAlex.
- Effect of Trinucleotide Repeats in the Huntington's Gene on Intelligence.EBioMedicine · 2018Trial
- Aberrant medial ganglionic eminence (MGE) GABAergic neurogenesis contributes to Huntington's disease pathogenesis.Neurobiology of disease · 2026Article
- Article
- Exploring huntington's disease from a neurodevelopmental perspective.International journal of biological sciences · 2026Review
- Brain organoid models of Huntington's disease shift the focus towards neurodevelopment.Disease models & mechanisms · 2025Article
- Distinct molecular patterns in R6/2 HD mouse brain: Insights from spatiotemporal transcriptomics.Neuron · 2025Article
- Cerulenin partially corrects the disrupted developmental transcriptomic signature in Huntington's disease striatal medium spiny neurons.Stem cells (Dayton, Ohio) · 2025Article
- HTT loss-of-function contributes to RNA deregulation in developing Huntington's disease neurons.Cell & bioscience · 2025Article
- The Role of MicroRNAs in Neurodegeneration: Insights from Huntington's Disease.Molecular neurobiology · 2025Review
- Molecular pathology, developmental changes and synaptic dysfunction in (pre-) symptomatic human C9ORF72-ALS/FTD cerebral organoids.Acta neuropathologica communications · 2024Article
- Research advances in huntingtin-associated protein 1 and its application prospects in diseases.Frontiers in neuroscience · 2024Review
- Genetic topography and cortical cell loss in Huntington's disease link development and neurodegeneration.Brain : a journal of neurology · 2023Article
- Unravelling the role of huntingtin: from neurodevelopment to neurodegeneration.Brain : a journal of neurology · 2023Article
- Gelation of cytoplasmic expanded CAG RNA repeats suppresses global protein synthesis.Nature chemical biology · 2023Article
- The C9ORF72 repeat expansion alters neurodevelopment.Cell reports · 2023Article
- The brain on time: links between development and neurodegeneration.Development (Cambridge, England) · 2023Article
- Transcriptional vulnerabilities of striatal neurons in human and rodent models of Huntington's disease.Nature communications · 2023Article
- Huntingtin-associated protein 1-associated intracellular trafficking in neurodegenerative diseases.Frontiers in aging neuroscience · 2023Review
- Synaptic Dysfunction in Huntington's Disease: Lessons from Genetic Animal Models.The Neuroscientist : a review journal bringing neurobiology, neurology and psychiatry · 2022Article
- Neurogenetic disorders across the lifespan: from aberrant development to degeneration.Nature reviews. Neurology · 2022Review
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Authors and funding
12 authors at 1 institution in 1 country.
Eduardo E Arteaga-BrachoRoslyn and Leslie Goldstein Laboratory for Stem Cell Biology and Regenerative Medicine, Albert Einstein College of Medicine, Bronx, NY, USA; Institute for Brain Disorders and Neural Regeneration, Albert Einstein College of Medicine, Bronx, NY, USA; Dominick P. Purpura Department of Neuroscience, Albert Einstein College of Medicine, Bronx, NY, USA; Rose F. Kennedy Center for Research on Intellectual and Developmental Disabilities, Albert Einstein College of Medicine, Bronx, NY, USA.
Maria GulinelloDominick P. Purpura Department of Neuroscience, Albert Einstein College of Medicine, Bronx, NY, USA; Rose F. Kennedy Center for Research on Intellectual and Developmental Disabilities, Albert Einstein College of Medicine, Bronx, NY, USA; Behavioral Core Facility, Albert Einstein College of Medicine, Bronx, NY, USA.
Michael L WinchesterRoslyn and Leslie Goldstein Laboratory for Stem Cell Biology and Regenerative Medicine, Albert Einstein College of Medicine, Bronx, NY, USA; Institute for Brain Disorders and Neural Regeneration, Albert Einstein College of Medicine, Bronx, NY, USA; The Saul R. Korey Department of Neurology, Albert Einstein College of Medicine, Bronx, NY, USA; Rose F. Kennedy Center for Research on Intellectual and Developmental Disabilities, Albert Einstein College of Medicine, Bronx, NY, USA.
Nandini PichamoorthyRoslyn and Leslie Goldstein Laboratory for Stem Cell Biology and Regenerative Medicine, Albert Einstein College of Medicine, Bronx, NY, USA; Institute for Brain Disorders and Neural Regeneration, Albert Einstein College of Medicine, Bronx, NY, USA; The Saul R. Korey Department of Neurology, Albert Einstein College of Medicine, Bronx, NY, USA; Rose F. Kennedy Center for Research on Intellectual and Developmental Disabilities, Albert Einstein College of Medicine, Bronx, NY, USA.
Jenna R PetrongloRoslyn and Leslie Goldstein Laboratory for Stem Cell Biology and Regenerative Medicine, Albert Einstein College of Medicine, Bronx, NY, USA; Institute for Brain Disorders and Neural Regeneration, Albert Einstein College of Medicine, Bronx, NY, USA; The Saul R. Korey Department of Neurology, Albert Einstein College of Medicine, Bronx, NY, USA; Rose F. Kennedy Center for Research on Intellectual and Developmental Disabilities, Albert Einstein College of Medicine, Bronx, NY, USA.
Alicia D ZambranoRoslyn and Leslie Goldstein Laboratory for Stem Cell Biology and Regenerative Medicine, Albert Einstein College of Medicine, Bronx, NY, USA; Institute for Brain Disorders and Neural Regeneration, Albert Einstein College of Medicine, Bronx, NY, USA; The Saul R. Korey Department of Neurology, Albert Einstein College of Medicine, Bronx, NY, USA.
Julio InocencioThe Saul R. Korey Department of Neurology, Albert Einstein College of Medicine, Bronx, NY, USA.
Chirstopher D De JesusRoslyn and Leslie Goldstein Laboratory for Stem Cell Biology and Regenerative Medicine, Albert Einstein College of Medicine, Bronx, NY, USA; Institute for Brain Disorders and Neural Regeneration, Albert Einstein College of Medicine, Bronx, NY, USA; Dominick P. Purpura Department of Neuroscience, Albert Einstein College of Medicine, Bronx, NY, USA; Rose F. Kennedy Center for Research on Intellectual and Developmental Disabilities, Albert Einstein College of Medicine, Bronx, NY, USA.
Joseph O LouieThe Saul R. Korey Department of Neurology, Albert Einstein College of Medicine, Bronx, NY, USA.
Solen GokhanRoslyn and Leslie Goldstein Laboratory for Stem Cell Biology and Regenerative Medicine, Albert Einstein College of Medicine, Bronx, NY, USA; Institute for Brain Disorders and Neural Regeneration, Albert Einstein College of Medicine, Bronx, NY, USA; The Saul R. Korey Department of Neurology, Albert Einstein College of Medicine, Bronx, NY, USA; Rose F. Kennedy Center for Research on Intellectual and Developmental Disabilities, Albert Einstein College of Medicine, Bronx, NY, USA.
Mark F MehlerRoslyn and Leslie Goldstein Laboratory for Stem Cell Biology and Regenerative Medicine, Albert Einstein College of Medicine, Bronx, NY, USA; Institute for Brain Disorders and Neural Regeneration, Albert Einstein College of Medicine, Bronx, NY, USA; The Saul R. Korey Department of Neurology, Albert Einstein College of Medicine, Bronx, NY, USA; Dominick P. Purpura Department of Neuroscience, Albert Einstein College of Medicine, Bronx, NY, USA; Department of Psychiatry and Behavioral Sciences, Albert Einstein College of Medicine, Bronx, NY, USA; Rose F. Kennedy Center for Research on Intellectual and Developmental Disabilities, Albert Einstein College of Medicine, Bronx, NY, USA; Einstein Cancer Center, Albert Einstein College of Medicine, Bronx, NY, USA; Ruth L. and David S. Gottesman Institute for Stem Cell Biology and Regenerative Medicine, Albert Einstein College of Medicine, Bronx, NY, USA; Center for Epigenomics, Albert Einstein College of Medicine, Bronx, NY, USA; Institute for Aging Research, Albert Einstein College of Medicine, Bronx, NY, USA.
Aldrin E MoleroRoslyn and Leslie Goldstein Laboratory for Stem Cell Biology and Regenerative Medicine, Albert Einstein College of Medicine, Bronx, NY, USA; Institute for Brain Disorders and Neural Regeneration, Albert Einstein College of Medicine, Bronx, NY, USA; The Saul R. Korey Department of Neurology, Albert Einstein College of Medicine, Bronx, NY, USA; Rose F. Kennedy Center for Research on Intellectual and Developmental Disabilities, Albert Einstein College of Medicine, Bronx, NY, USA. Electronic address: aldrin.molero@einstein.yu.edu.
Albert Einstein College of Medicine · US
Funding
Translational Neuroimaging CoreP30HD071593 · NICHD · ALBERT EINSTEIN COLLEGE OF MEDICINE, INC · PI WALKLEY, STEVEN UPSHAW · 2011 to 2015
$5.6MHuntington's disease: a novel developmental oligodendrogliopathyR01NS096144 · NINDS · ALBERT EINSTEIN COLLEGE OF MEDICINE, INC · PI MEHLER, MARK F · 2016 to 2020
$1.8MThe Role of Huntingtin in Striatal NeurogenesisR01NS073758 · NINDS · ALBERT EINSTEIN COLLEGE OF MEDICINE, INC · PI MOLERO, ALDRIN ENRIQUE · 2011 to 2015
$1.6MNeurodegenerative Diseases: A New Class of Primary Developmental Disorders?R01NS071571 · NINDS · ALBERT EINSTEIN COLLEGE OF MEDICINE, INC · PI MEHLER, MARK F · 2010 to 2013
$1.3MNICHD NIH HHS P30 HD071593NINDS NIH HHS R01 NS071571NINDS NIH HHS R01 NS073758NINDS NIH HHS R01 NS096144
Abstract
The mutation in huntingtin (mHtt) leads to a spectrum of impairments in the developing forebrain of Huntington's disease (HD) mouse models. Whether these developmental alterations are due to loss- or gain-of-function mechanisms and contribute to HD pathogenesis is unknown. We examined the role of selective loss of huntingtin (Htt) function during development on postnatal vulnerability to cell death. We employed mice expressing very low levels of Htt throughout embryonic life to postnatal day 21 (Hdh
Indexed as
Age FactorsAnimalsAnimals, NewbornCell DifferentiationDevelopmental DisabilitiesDisease Models, AnimalEmbryo, MammalianGene Expression Regulation, DevelopmentalHuntingtin ProteinHuntington DiseaseMiceMice, TransgenicMutationNerve Tissue ProteinsNeurodegenerative DiseasesPsychomotor DisordersHuntingtin ProteinNerve Tissue ProteinsRNA, MessengerDegenerationDevelopmentGliogenesisHuntingtinHuntington's diseaseLoss-of-functionMaturationMyelinNeurogenesisPathogenesis
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.