{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,7]],"date-time":"2026-10-07T06:40:03Z","timestamp":1791355203982,"version":"4.3.1"},"reference-count":48,"publisher":"Wiley","issue":"23","license":[{"start":{"date-parts":[[2023,10,21]],"date-time":"2023-10-21T00:00:00Z","timestamp":1697846400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["physoc.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["The Journal of Physiology"],"published-print":{"date-parts":[[2023,12]]},"abstract":"\n Abstract<\/jats:title>\n \n In\n \n Nyx\n nob<\/jats:sup>\n <\/jats:italic>\n mice, a model for congenital nystagmus associated with congenital stationary night blindness (CSNB), synchronous oscillating retinal ganglion cells (RGCs) lead to oscillatory eye movements, i.e. nystagmus. Given the specific expression of\n mGluR6<\/jats:italic>\n and\n \n Ca\n v<\/jats:sub>\n 1.4\n <\/jats:italic>\n in the photoreceptor to bipolar cell synapses, as well as their clinical association with CSNB, we hypothesize that\n \n Grm6\n nob3<\/jats:sup>\n <\/jats:italic>\n and\n \n Ca\n v<\/jats:sub>\n 1.4\u2010KO\n <\/jats:italic>\n mutants show, like the\n \n Nyx\n nob<\/jats:sup>\n <\/jats:italic>\n mouse, oscillations in both their RGC activity and eye movements. Using multi\u2010electrode array recordings of RGCs and measurements of the eye movements, we demonstrate that\n \n Grm6\n nob3<\/jats:sup>\n <\/jats:italic>\n and\n \n Ca\n v<\/jats:sub>\n 1.4\u2010KO\n <\/jats:italic>\n mice also show oscillations of their RGCs as well as a nystagmus. Interestingly, the preferred frequencies of RGC activity as well as the eye movement oscillations of the\n \n Grm6\n nob3<\/jats:sup>\n <\/jats:italic>\n ,\n \n Ca\n v<\/jats:sub>\n 1.4\u2010KO\n <\/jats:italic>\n and\n \n Nyx\n nob<\/jats:sup>\n <\/jats:italic>\n mice differ among mutants, but the neuronal activity and eye movement behaviour within a strain remain aligned in the same frequency domain. Model simulations indicate that mutations affecting the photoreceptor\u2013bipolar cell synapse can form a common cause of the nystagmus of CSNB by driving oscillations in RGCs via A\n II<\/jats:sub>\n amacrine cells.\n \n \n image<\/jats:alt-text>\n <\/jats:graphic>\n <\/jats:boxed-text>\n <\/jats:p>\n <\/jats:sec>\n \n Key points<\/jats:title>\n \n \n \n \n In\n \n Nyx\n nob<\/jats:sup>\n <\/jats:italic>\n mice, a model for congenital nystagmus associated with congenital stationary night blindness (CSNB), their oscillatory eye movements (i.e. nystagmus) are caused by synchronous oscillating retinal ganglion cells.\n <\/jats:p>\n <\/jats:list-item>\n \n \n Here we show that the same mechanism applies for two other CSNB mouse models \u2013\n \n Grm6\n nob3<\/jats:sup>\n <\/jats:italic>\n and\n \n Ca\n v<\/jats:sub>\n 1.4\u2010KO\n <\/jats:italic>\n mice.\n <\/jats:p>\n <\/jats:list-item>\n \n \n We propose that the retinal ganglion cell oscillations originate in the A\n II<\/jats:sub>\n amacrine cells.\n <\/jats:p>\n <\/jats:list-item>\n \n Model simulations show that by only changing the input to ON\u2010bipolar cells, all phenotypical differences between the various genetic mouse models can be reproduced.<\/jats:p>\n <\/jats:list-item>\n <\/jats:list>\n <\/jats:p>\n <\/jats:sec>","DOI":"10.1113\/jp284965","type":"journal-article","created":{"date-parts":[[2023,10,21]],"date-time":"2023-10-21T09:09:21Z","timestamp":1697879361000},"page":"5317-5340","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["A common cause for nystagmus in different congenital stationary night blindness mouse models"],"prefix":"10.1113","volume":"601","author":[{"given":"Maj\u2010Britt","family":"H\u00f6lzel","sequence":"first","affiliation":[{"name":"Netherlands Institute for Neuroscience Amsterdam Amsterdam the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wouter","family":"Kamermans","sequence":"additional","affiliation":[{"name":"Netherlands Institute for Neuroscience Amsterdam Amsterdam the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Beerend H. 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