{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,12]],"date-time":"2026-03-12T09:41:59Z","timestamp":1773308519222,"version":"3.50.1"},"reference-count":35,"publisher":"SAGE Publications","issue":"2","license":[{"start":{"date-parts":[[2023,4,1]],"date-time":"2023-04-01T00:00:00Z","timestamp":1680307200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Zebrafish"],"published-print":{"date-parts":[[2023,4,1]]},"abstract":"<jats:p>\n                    Our understanding of inner ear hair cell ultrastructure has heretofore relied upon two-dimensional imaging; however, serial block-face scanning electron microscopy (SBFSEM) changes this paradigm allowing for three-dimensional evaluation. We compared inner ear hair cells of the apical cristae in\n                    <jats:italic toggle=\"yes\">\n                      myo7aa\n                      <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    <\/jats:italic>\n                    null zebrafish, a model of human Usher Syndrome type 1B, to hair cells in wild-type zebrafish by SBFSEM to investigate possible ribbon synapse ultrastructural differences. Previously, it has been shown that compared to wild type,\n                    <jats:italic toggle=\"yes\">\n                      myo7aa\n                      <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    <\/jats:italic>\n                    zebrafish neuromast hair cells have fewer ribbon synapses yet similar ribbon areas. We expect the recapitulation of these results within the inner ear apical crista hair cells furthering the knowledge of three-dimensional ribbon synapse structure while resolving the feasibility of therapeutically targeting\n                    <jats:italic toggle=\"yes\">\n                      myo7aa\n                      <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    <\/jats:italic>\n                    mutant ribbons. In this report, we evaluated ribbon synapse number, volume, surface area, and sphericity. Localization of ribbons and their distance from the nearest innervation were also evaluated. We determined that\n                    <jats:italic toggle=\"yes\">\n                      myo7aa\n                      <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    <\/jats:italic>\n                    mutant ribbon synapses are smaller in volume and surface area; however, all other measurements were not significantly different from wild-type zebrafish. Because the ribbon synapses are nearly indistinguishable between the\n                    <jats:italic toggle=\"yes\">\n                      myo7aa\n                      <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    <\/jats:italic>\n                    mutant and wild type, it suggests that the ribbons are structurally receptive, supporting that therapeutic intervention may be feasible.\n                  <\/jats:p>","DOI":"10.1089\/zeb.2022.0049","type":"journal-article","created":{"date-parts":[[2023,4,18]],"date-time":"2023-04-18T16:25:03Z","timestamp":1681835103000},"page":"47-54","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":1,"title":["Three-Dimensional Structure of Inner Ear Hair Cell Ribbon Synapses in a Zebrafish Model of Usher Syndrome Type 1B"],"prefix":"10.1177","volume":"20","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5981-682X","authenticated-orcid":false,"suffix":"Jr.","given":"Kenneth C.","family":"Riley","sequence":"first","affiliation":[{"name":"Department of Clinical Genomics, Mayo Clinic, Rochester, Minnesota, USA."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7949-7205","authenticated-orcid":false,"given":"Alaa","family":"Koleilat","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, Minnesota, USA."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4324-7658","authenticated-orcid":false,"given":"Joseph A.","family":"Dugdale","sequence":"additional","affiliation":[{"name":"Department of Otorhinolaryngology, Head and Neck Surgery, Mayo Clinic, Rochester, Minnesota, USA."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2669-7275","authenticated-orcid":false,"given":"Shawna A.","family":"Cooper","sequence":"additional","affiliation":[{"name":"Mayo Clinic Graduate School of Biomedical Sciences, Mayo Clinic, Rochester, Minnesota, USA."},{"name":"Department of Biochemistry and Molecular Biology, Mayo Clinic, Rochester, Minnesota, USA."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4862-6259","authenticated-orcid":false,"given":"Trace A.","family":"Christensen","sequence":"additional","affiliation":[{"name":"Department of Biochemistry and Molecular Biology, Mayo Clinic, Rochester, Minnesota, USA."},{"name":"Microscopy and Cell Analysis Core, and Mayo Clinic, Rochester, Minnesota, USA."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3154-4249","authenticated-orcid":false,"given":"Lisa A.","family":"Schimmenti","sequence":"additional","affiliation":[{"name":"Department of Clinical Genomics, Mayo Clinic, Rochester, Minnesota, USA."},{"name":"Department of Otorhinolaryngology, Head and Neck Surgery, Mayo Clinic, Rochester, Minnesota, USA."},{"name":"Department of Biochemistry and Molecular Biology, Mayo Clinic, Rochester, Minnesota, USA."},{"name":"Department of Ophthalmology, Mayo Clinic, Rochester, Minnesota, USA."}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"179","published-online":{"date-parts":[[2023,4,13]]},"reference":[{"key":"e_1_3_6_2_2","doi-asserted-by":"publisher","DOI":"10.1146\/annurev.genet.39.073003.105049"},{"key":"e_1_3_6_3_2","doi-asserted-by":"publisher","DOI":"10.1016\/bs.mcb.2016.02.007"},{"key":"e_1_3_6_4_2","doi-asserted-by":"publisher","DOI":"10.1242\/jeb.087635"},{"key":"e_1_3_6_5_2","doi-asserted-by":"publisher","DOI":"10.1242\/jeb.01534"},{"key":"e_1_3_6_6_2","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/9.14.2189"},{"key":"e_1_3_6_7_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0896-6273(00)80455-9"},{"key":"e_1_3_6_8_2","unstructured":"Koenekoop RK Arriaga MA Trzupek KM Lentz JJ. 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