{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T01:37:00Z","timestamp":1760060220648,"version":"build-2065373602"},"reference-count":36,"publisher":"MDPI AG","issue":"3","license":[{"start":{"date-parts":[[2025,8,18]],"date-time":"2025-08-18T00:00:00Z","timestamp":1755475200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Wooten Family Initiative for Brain Health Research grant","award":["R15GM129679"],"award-info":[{"award-number":["R15GM129679"]}]},{"name":"National Institutes of Health (NIH)","award":["R15GM129679"],"award-info":[{"award-number":["R15GM129679"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["IJTM"],"abstract":"<jats:p>Background\/Objectives: Perturbation in terminal N-glycan processing is a feature of congenital disorders of glycosylation and neurological disorders. Since treatment options are limited, N-glycans are plausible therapeutic targets. Here, we investigated the consequences of substituting complex\/hybrid with oligomannose types of N-glycans on nervous and musculature systems, employing mgat1a and mgat1b mutant zebrafish models. Methods: CRISPR Cas9 technology was employed to engineer the mgat1a zebrafish model. The N-glycan populations in Wt AB, mgat1a\u2212\/\u2212 and mgat1b\u2212\/\u2212 zebrafish were characterized via lectin blotting. Motor and sensory functions were measured by tail-coiling and touch-evoked response assays in embryos and larvae. Swimming locomotion and anxiety-like behavior were characterized in adult Wt AB, and mutant zebrafish using motility and novel tank dive assays. Results: The mgat1a\u2212\/\u2212 model had increased oligomannosylated proteins compared to Wt AB in embryos and dissected brain, spinal cord, skeletal muscle, heart, swim bladder, and skin from adults, supporting a global knockdown of GnT-I activity. Higher levels were also observed in mgat1a\u2212\/\u2212 relative to mgat1b\u2212\/\u2212, except in the brain. Band patterns for oligomannosylated proteins were different between all three zebrafish lines. The mgat1\u2212\/\u2212 embryos and larvae had deficient motor and sensory functions which persisted into adulthood, with a higher deficiency in mgat1b\u2212\/\u2212. Anxiety-like behavior was decreased and increased in adult mgat1a\u2212\/\u2212 and mgat1b\u2212\/\u2212, respectively, compared to Wt AB. Conclusions: Taken together, this study revealed that aberrant terminal N-glycan processing impacts brain, spinal and muscle control, and hence will enhance our understanding of the vital role of complex\/hybrid N-glycans in nervous system health.<\/jats:p>","DOI":"10.3390\/ijtm5030036","type":"journal-article","created":{"date-parts":[[2025,8,18]],"date-time":"2025-08-18T13:28:22Z","timestamp":1755523702000},"page":"36","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Comparison of the Differing Impacts of Lowered N-Acetylglucosaminyltransferase-Ia\/b Activity on Motor and Sensory Function in Zebrafish"],"prefix":"10.3390","volume":"5","author":[{"ORCID":"https:\/\/orcid.org\/0009-0009-4065-7369","authenticated-orcid":false,"given":"M. Kristen","family":"Hall","sequence":"first","affiliation":[{"name":"Department of Biochemistry and Molecular Biology, Brody School of Medicine, East Carolina University, Greenville, NC 27834, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4595-3425","authenticated-orcid":false,"given":"Cody J.","family":"Hatchett","sequence":"additional","affiliation":[{"name":"Department of Biochemistry and Molecular Biology, Brody School of Medicine, East Carolina University, Greenville, NC 27834, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Haris A.","family":"Khan","sequence":"additional","affiliation":[{"name":"Department of Biochemistry and Molecular Biology, Brody School of Medicine, East Carolina University, Greenville, NC 27834, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hannah","family":"Lewis","sequence":"additional","affiliation":[{"name":"Department of Biochemistry and Molecular Biology, Brody School of Medicine, East Carolina University, Greenville, NC 27834, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6839-7863","authenticated-orcid":false,"given":"Ruth A.","family":"Schwalbe","sequence":"additional","affiliation":[{"name":"Department of Biochemistry and Molecular Biology, Brody School of Medicine, East Carolina University, Greenville, NC 27834, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2025,8,18]]},"reference":[{"key":"ref_1","unstructured":"Varki, A., Cummings, R.D., Esko, J.D., Stanley, P., Hart, G.W., Aebi, M., Mohnen, D., Kinoshita, T., Packer, N.H., and Prestegard, J.H. 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