ArticleThe Journal of biological chemistry2013
Zebrafish as a model for monocarboxyl transporter 8-deficiency.
Article in The Journal of biological chemistry, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 35 papers.
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Who cites it
35 citing papers in PubMed, 68 citations in OpenAlex.
- Experimentally Manipulating the Thyroid Hormone Axis in Zebrafish.Methods in molecular biology (Clifton, N.J.) · 2025Review
- Cross-species applicability of an adverse outcome pathway network for thyroid hormone system disruption.Toxicological sciences : an official journal of the Society of Toxicology · 2023Review
- Development and metamorphosis in frogs deficient in the thyroid hormone transporter MCT8.General and comparative endocrinology · 2023Article
- In a zebrafish biomedical model of human Allan-Herndon-Dudley syndrome impaired MTH signaling leads to decreased neural cell diversity.Frontiers in endocrinology · 2023Article
- AAV9-MCT8 Delivery at Juvenile Stage Ameliorates Neurological and Behavioral Deficits in a Mouse Model of MCT8-Deficiency.Thyroid : official journal of the American Thyroid Association · 2022Article
- Cardiac forces regulate zebrafish heart valve delamination by modulating Nfat signaling.PLoS biology · 2022Article
- Monocarboxylate Transporter 8 Deficiency: From Pathophysiological Understanding to Therapy Development.Frontiers in endocrinology · 2021Review
- Thyroid hormone regulation of neural stem cell fate: From development to ageing.Acta physiologica (Oxford, England) · 2020Review
- The Role of the Thyroid Axis in Fish.Frontiers in endocrinology · 2020Review
- Monocarboxylate Transporter 8 Deficiency: Delayed or Permanent Hypomyelination?Frontiers in endocrinology · 2020Review
- MCT8 Deficiency: The Road to Therapies for a Rare Disease.Frontiers in neuroscience · 2020Review
- Changes in thyroid hormone activity disrupt photomotor behavior of larval zebrafish.Neurotoxicology · 2019Article
- Effects of thyroid hormone disruption on the ontogenetic expression of thyroid hormone signaling genes in developing zebrafish (Danio rerio).General and comparative endocrinology · 2019Article
- Thyroid Hormone Hyposensitivity: From Genotype to Phenotype and Back.Frontiers in endocrinology · 2019Review
- Gene transcription ontogeny of hypothalamic-pituitary-thyroid axis development in early-life stage fathead minnow and zebrafish.General and comparative endocrinology · 2018Article
- Thyroid hormone and the brain: Mechanisms of action in development and role in protection and promotion of recovery after brain injury.Pharmacology & therapeutics · 2018Review
- Transcriptomics reveal an integrative role for maternal thyroid hormones during zebrafish embryogenesis.Scientific reports · 2017Article
- Sleep-Dependent Structural Synaptic Plasticity of Inhibitory Synapses in the Dendrites of Hypocretin/Orexin Neurons.Molecular neurobiology · 2017Article
- Modeling Psychomotor Retardation using iPSCs from MCT8-Deficient Patients Indicates a Prominent Role for the Blood-Brain Barrier.Cell stem cell · 2017Article
- New insights into thyroid hormone action.Pharmacology & therapeutics · 2017Review
Corrections and comments
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Authors and funding
7 authors at 2 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Allan-Herndon-Dudley syndrome (AHDS) is a severe psychomotor retardation characterized by neurological impairment and abnormal thyroid hormone (TH) levels. Mutations in the TH transporter, monocarboxylate transporter 8 (MCT8), are associated with AHDS. MCT8 knock-out mice exhibit impaired TH levels; however, they lack neurological defects. Here, the zebrafish mct8 gene and promoter were isolated, and mct8 promoter-driven transgenic lines were used to show that, similar to humans, mct8 is primarily expressed in the nervous and vascular systems. Morpholino-based knockdown and rescue experiments revealed that MCT8 is strictly required for neural development in the brain and spinal cord. This study shows that MCT8 is a crucial regulator during embryonic development and establishes the first vertebrate model for MCT8 deficiency that exhibits a neurological phenotype.
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Registered trials
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.