ArticleDisease models & mechanisms2016
Pharmacological treatment and BBB-targeted genetic therapy for MCT8-dependent hypomyelination in zebrafish.
Article in Disease models & mechanisms, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 37 papers, 1 of them a synthesis that pooled it.
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The trial behind it
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Who cites it
37 citing papers in PubMed, 1 synthesis or guideline pooled it, 65 citations in OpenAlex.
- Pooled it
- Effectiveness and safety of the tri-iodothyronine analogue Triac in children and adults with MCT8 deficiency: an international, single-arm, open-label, phase 2 trial.The lancet. Diabetes & endocrinology · 2019Trial
- Reduced thyroid hormone transport in a human placental model with inhibited MCT8.European thyroid journal · 2026Article
- A cost-based-plus pricing approach for repurposed tiratricol in the treatment of Allan-Herndon-Dudley syndrome.Orphanet journal of rare diseases · 2026Article
- Evaluating the effects of pro-myelinating drugs on motor function and myelination in a zebrafish model of genetic leukoencephalopathy.Neuroscience letters · 2025Article
- Mapping variants in thyroid hormone transporter MCT8 to disease severity by genomic, phenotypic, functional, structural and deep learning integration.Nature communications · 2025Article
- Sox10 is required for systemic initiation of bone mineralization.Development (Cambridge, England) · 2025Article
- Experimentally Manipulating the Thyroid Hormone Axis in Zebrafish.Methods in molecular biology (Clifton, N.J.) · 2025Review
- Diagnosis and Therapy in MCT8 Deficiency: Ongoing ChallengesJournal of clinical research in pediatric endocrinology · 2024Article
- Impaired T3 uptake and action in MCT8-deficient cerebral organoids underlie Allan-Herndon-Dudley syndrome.JCI insight · 2024Article
- Diving deep: zebrafish models in motor neuron degeneration research.Frontiers in neuroscience · 2024Review
- Imaging Approaches to Investigate Pathophysiological Mechanisms of Brain Disease in Zebrafish.International journal of molecular sciences · 2023Review
- Article
- Triac Treatment Prevents Neurodevelopmental and Locomotor Impairments in Thyroid Hormone Transporter Mct8/Oatp1c1 Deficient Mice.International journal of molecular sciences · 2023Article
- Tsh Induces Agrp1 Neuron Proliferation in Oatp1c1-Deficient Zebrafish.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2022Article
- Genetic disorders of thyroid development, hormone biosynthesis and signalling.Clinical endocrinology · 2022Review
- Article
- Review
- Genetic and Neurological Deficiencies in the Visual System ofInternational journal of molecular sciences · 2022Article
- Long-Term Efficacy of T3 Analogue Triac in Children and Adults With MCT8 Deficiency: A Real-Life Retrospective Cohort Study.The Journal of clinical endocrinology and metabolism · 2022Article
Corrections and comments
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Authors and funding
4 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Hypomyelination is a key symptom of Allan-Herndon-Dudley syndrome (AHDS), a psychomotor retardation associated with mutations in the thyroid-hormone (TH) transporter MCT8 (monocarboxylate transporter 8). AHDS is characterized by severe intellectual deficiency, neuromuscular impairment and brain hypothyroidism. In order to understand the mechanism for TH-dependent hypomyelination, we developed an mct8 mutant (mct8
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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.