{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,11,7]],"date-time":"2026-11-07T20:29:32Z","timestamp":1794083372578,"version":"4.8.0"},"reference-count":39,"publisher":"Wiley","issue":"9","license":[{"start":{"date-parts":[[2017,4,24]],"date-time":"2017-04-24T00:00:00Z","timestamp":1492992000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Developmental Neurobiology"],"published-print":{"date-parts":[[2017,9]]},"abstract":"<jats:title>ABSTRACT<\/jats:title><jats:p>Rett syndrome is a severe neurodevelopmental disorder. It is caused by a mutation in methyl\u2010CpG binding protein 2 (MecP2), a transcriptional regulator that recruits protein complexes involved in histone modification and chromatin remodeling. However, the role of Mecp2 in Rett syndrome remains unclear. In this study, we investigated the function of Mecp2 in neuronal development using zebrafish embryos. Mecp2 expression was detected ubiquitously in the central nervous system and muscles at 28 h postfertilization (hpf). We injected an antisense morpholino oligonucleotide (AMO) to induce Mecp2 knockdown phenotype. In <jats:italic>mecp2<\/jats:italic> morphants (embryos with Mecp2 knockdown by AMO) at 28 and 72 hpf, we found an increase in abnormal axonal branches of caudal primary motor neurons and a decrease in motor activity. In <jats:italic>mecp2<\/jats:italic> morphants at 24 hpf, we observed an increase in the expression of an <jats:italic>mecp2<\/jats:italic> downstream candidate gene, <jats:italic>brain derived neurotrophic factor<\/jats:italic> (<jats:italic>bdnf)<\/jats:italic>. In <jats:italic>mecp2<\/jats:italic> morphants at 72 hpf, the presynaptic area stained by an anti\u2010SV2 antibody was increased at the neuromuscular junction (NMJ). Interestingly, the size of SV2\u2010positive presynaptic area at the NMJ was also increased following <jats:italic>bdnf<\/jats:italic> mRNA injection, while it was normalized in a double knockdown of <jats:italic>mecp2<\/jats:italic> and <jats:italic>bdnf<\/jats:italic>. These results imply that Mecp2 is an important functional regulator of <jats:italic>bdnf<\/jats:italic> gene expression during neural circuit formation in zebrafish embryo. \u00a9 2017 Wiley Periodicals, Inc. 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