ArticleHuman molecular genetics2016
Attenuated pupillary light responses and downregulation of opsin expression parallel decline in circadian disruption in two different mouse models of Huntington's disease.
Article in Human molecular genetics, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 16 papers.
What it found
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
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Who cites it
16 citing papers in PubMed, 25 citations in OpenAlex.
- The Retinal Dopaminergic Circuit as a Biomarker for Huntington's and Alzheimer's Diseases.International journal of molecular sciences · 2025Article
- Preservation of Intrinsically Photosensitive Retinal Ganglion Cells (ipRGCs) in Late Adult Mice: Implications as a Potential Biomarker for Early Onset Ocular Degenerative Diseases.Investigative ophthalmology & visual science · 2024Article
- Increase in brain glycogen levels ameliorates Huntington's disease phenotype and rescues neurodegeneration in Drosophila.Disease models & mechanisms · 2023Article
- Sleep Disorders and Circadian Disruption in Huntington's Disease.Journal of Huntington's disease · 2023Review
- Sleep and Circadian Rhythm Dysfunction in Animal Models of Huntington's Disease.Journal of Huntington's disease · 2023Review
- A New Perspective on Huntington's Disease: How a Neurological Disorder Influences the Peripheral Tissues.International journal of molecular sciences · 2022Review
- Autonomic disorders and myocardial 123I-metaiodobenzylguanidine scintigraphy in Huntington's disease.Journal of nuclear cardiology : official publication of the American Society of Nuclear Cardiology · 2022Article
- Circadian rhythms in neurodegenerative disorders.Nature reviews. Neurology · 2022Review
- Defining circadian disruption in neurodegenerative disorders.The Journal of clinical investigation · 2021Review
- Circadian dysfunction in the Q175 model of Huntington's disease: Network analysis.Journal of neuroscience research · 2019Article
- Degeneration of ipRGCs in Mouse Models of Huntington's Disease Disrupts Non-Image-Forming Behaviors Before Motor Impairment.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2019Article
- Differential roles for cryptochromes in the mammalian retinal clock.FASEB journal : official publication of the Federation of American Societies for Experimental Biology · 2018Article
- Melanopsin Retinal Ganglion Cells and Pupil: Clinical Implications for Neuro-Ophthalmology.Frontiers in neurology · 2018Review
- Prolonged day length exposure improves circadian deficits and survival in a transgenic mouse model of Huntington's disease.Neurobiology of sleep and circadian rhythms · 2017Article
- Retinal Ganglion Cells and Circadian Rhythms in Alzheimer's Disease, Parkinson's Disease, and Beyond.Frontiers in neurology · 2017Review
- Article
Corrections and comments
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Authors and funding
5 authors at 2 institutions in 1 country.
Funding
Abstract
Circadian deficits in Huntington's disease (HD) are recapitulated in both fragment (R6/2) and full-length (Q175) mouse models of HD. Circadian rhythms are regulated by the suprachiasmatic nuclei (SCN) in the hypothalamus, which are primarily entrained by light detected by the retina. The SCN receives input from intrinsically photosensitive retinal ganglion cells (ipRGCs) that express the photopigment melanopsin, but also receive input from rods and cones. In turn, ipRGCs mediate a range of non-image forming responses to light including circadian entrainment and the pupillary light response (PLR). Retinal degeneration/dysfunction has been described previously in R6/2 mice. We investigated, therefore, whether or not circadian disruption in HD mice is due to abnormalities in retinal photoreception. We measured the expression of melanopsin, rhodopsin and cone opsin, as well as other retinal markers (tyrosine hydroxylase, calbindin, PKCα and Brna3), in R6/2 and Q175 mice at different stages of disease. We also measured the PLR as a 'readout' for ipRGC function and a marker of light reception by the retina. We found that the PLR was attenuated in both lines of HD mice. This was accompanied by a progressive downregulation of cone opsin and melanopsin expression. We suggest that disease-related changes in photoreception by the retina contribute to the progressive dysregulation of circadian rhythmicity and entrainment seen in HD mice. Colour vision is abnormal in HD patients. Therefore, if retinal deficits similar to those seen in HD mice are confirmed in patients, specifically designed light therapy may be an effective strategy to improve circadian dysfunction.
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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.