ArticlePLoS biology2019
Nystagmus in patients with congenital stationary night blindness (CSNB) originates from synchronously firing retinal ganglion cells.
Article in PLoS biology, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers, 1 of them a synthesis that pooled it.
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Who cites it
18 citing papers in PubMed, 1 synthesis or guideline pooled it, 34 citations in OpenAlex.
- Symptomatic treatment of infantile nystagmus: a systematic review.Frontiers in neuroscience · 2025Pooled it
- Dopamine suppresses pathological retinal oscillations and enhances the signal-to-noise ratio.Proceedings of the National Academy of Sciences of the United States of America · 2026Article
- Diversity and Feature Selectivity of Primate Retinal Ganglion Cells.Annual review of vision science · 2026Review
- Gene augmentation therapy successfully treats mice with complete congenital stationary night blindness (cCSNB), improving retinal function and visual acuity.Gene therapy · 2026Article
- A mechanism for pathological oscillations in mouse retinal ganglion cells in a model of night blindness.The Journal of general physiology · 2025Article
- A Novel Type of Autosomal Dominant Episodic Nystagmus Segregating with a Variant in the FRMD5 Gene.Neuro-ophthalmology (Aeolus Press) · 2024Article
- Article
- Impaired Direction Selectivity in the Nucleus of the Optic Tract of Albino Mice.Investigative ophthalmology & visual science · 2023Article
- LRIT3 expression in cone photoreceptors restores post-synaptic bipolar cell signalplex assembly and partial function iniScience · 2023Article
- Differences in visual stimulation between reading and walking and implications for myopia development.Journal of vision · 2023Article
- A retinal origin of nystagmus-a perspective.Frontiers in ophthalmology · 2023Article
- Impaired Extraocular Muscle Innervation Is Present Before Eye Opening in a Mouse Model of Infantile Nystagmus Syndrome.Investigative ophthalmology & visual science · 2022Article
- ON than OFF pathway disruption leads to greater deficits in visual function and retinal dopamine signaling.Experimental eye research · 2022Article
- Nystagmus only with fixation in the light: a rare central sign due to cerebellar malfunction.Journal of neurology · 2022Article
- Conserved circuits for direction selectivity in the primate retina.Current biology : CB · 2022Article
- Receptive Field Sizes ofInternational journal of molecular sciences · 2022Article
- SLC38A8 mutations result in arrested retinal development with loss of cone photoreceptor specialization.Human molecular genetics · 2020Article
- p.His16Arg of STXBP1 (MUNC18-1) Associated With Syntaxin 3B Causes Autosomal Dominant Congenital Nystagmus.Frontiers in cell and developmental biology · 2020Article
Corrections and comments
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Authors and funding
13 authors at 3 institutions in 3 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Congenital nystagmus, involuntary oscillating small eye movements, is commonly thought to originate from aberrant interactions between brainstem nuclei and foveal cortical pathways. Here, we investigated whether nystagmus associated with congenital stationary night blindness (CSNB) results from primary deficits in the retina. We found that CSNB patients as well as an animal model (nob mice), both of which lacked functional nyctalopin protein (NYX, nyx) in ON bipolar cells (BCs) at their synapse with photoreceptors, showed oscillating eye movements at a frequency of 4-7 Hz. nob ON direction-selective ganglion cells (DSGCs), which detect global motion and project to the accessory optic system (AOS), oscillated with the same frequency as their eyes. In the dark, individual ganglion cells (GCs) oscillated asynchronously, but their oscillations became synchronized by light stimulation. Likewise, both patient and nob mice oscillating eye movements were only present in the light when contrast was present. Retinal pharmacological and genetic manipulations that blocked nob GC oscillations also eliminated their oscillating eye movements, and retinal pharmacological manipulations that reduced the oscillation frequency of nob GCs also reduced the oscillation frequency of their eye movements. We conclude that, in nob mice, synchronized oscillations of retinal GCs, most likely the ON-DCGCs, cause nystagmus with properties similar to those associated with CSNB in humans. These results show that the nob mouse is the first animal model for a form of congenital nystagmus, paving the way for development of therapeutic strategies.
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