{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,24]],"date-time":"2026-07-24T21:26:37Z","timestamp":1784928397464,"version":"3.55.0"},"reference-count":48,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2016,6,1]],"date-time":"2016-06-01T00:00:00Z","timestamp":1464739200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100001659","name":"Deutsche Forschungsgemeinschaft","doi-asserted-by":"publisher","award":["GRK1885\/1"],"award-info":[{"award-number":["GRK1885\/1"]}],"id":[{"id":"10.13039\/501100001659","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Eur J of Neuroscience"],"published-print":{"date-parts":[[2016,7]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Complex sphingolipids are strongly expressed in neuronal tissue and contain ceramides in their backbone. Ceramides are synthesized by six ceramide synthases (CerS1\u20136). Although it is known that each tissue has a unique profile of ceramide synthase expression and ceramide synthases are implicated in several neurodegenerative disorders, the expression of ceramide synthase isoforms has not been investigated in the retina. Here we demonstrate CerS1, CerS2 and CerS4 expression in mouse retina and cornea, with CerS4 ubiquitously expressed in all retinal neurons and M\u00fcller cells. To test whether ceramide synthase deficiency affects retinal function, we compared electroretinograms and retina morphology between wild\u2010type and CerS1\u2010, CerS2\u2010 and CerS4\u2010deficient mice. Electroretinograms were strongly reduced in amplitude in ceramide synthase\u2010deficient mice, suggesting that signalling in the outer retina is affected. However, the number of photoreceptors and cone outer segment length were unaltered and no changes in retinal layer thickness or synaptic structures were found. Mass spectrometric analyses of ceramides, hexosyl\u2010ceramides and sphingomyelins showed that C20 to C24 acyl\u2010containing species were decreased whereas C16\u2010containing species were increased in the retina of ceramide synthase\u2010deficient mice. Similar but smaller changes were also found in the cornea. Thus, we hypothesize that the replacement of very long\u2010chain fatty acyl residues by shorter C16 residues may affect the electrical properties of retina and cornea, and alter receptor\u2010mediated signal transduction, vesicle\u2010mediated synaptic transmission or corneal light transmission. Future studies need to identify the molecular targets of ceramides or derived sphingolipids in light signal transduction and transmission in the eye.<\/jats:p>","DOI":"10.1111\/ejn.13260","type":"journal-article","created":{"date-parts":[[2016,4,18]],"date-time":"2016-04-18T08:43:59Z","timestamp":1460969039000},"page":"1700-1713","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":14,"title":["Defective ceramide synthases in mice cause reduced amplitudes in electroretinograms and altered sphingolipid composition in retina and cornea"],"prefix":"10.1111","volume":"44","author":[{"given":"Bianca","family":"Br\u00fcggen","sequence":"first","affiliation":[{"name":"Neurobiology University of Oldenburg 26111 Oldenburg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christiane","family":"Kremser","sequence":"additional","affiliation":[{"name":"Life and Medical Sciences Institute University of Bonn Bonn 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