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Several studies have linked the loss of MeCP2 function to alterations of brain\u2010derived neurotrophic factor (BDNF) levels, but non\u2010specific overexpression of BDNF only partially improves the phenotype of\n                    <jats:italic>Mecp2<\/jats:italic>\n                    \u2010deficient mice. We and others have previously shown that huntingtin (HTT) scaffolds molecular motor complexes, transports BDNF\u2010containing vesicles, and is under\u2010expressed in\n                    <jats:italic>Mecp2<\/jats:italic>\n                    knockout brains. Here, we demonstrate that promoting HTT phosphorylation at Ser421, either by a phospho\u2010mimetic mutation or inhibition of the phosphatase calcineurin, restores endogenous BDNF axonal transport\n                    <jats:italic>in\u00a0vitro<\/jats:italic>\n                    in the corticostriatal pathway, increases striatal BDNF availability and synaptic connectivity\n                    <jats:italic>in\u00a0vivo,<\/jats:italic>\n                    and improves the phenotype and the survival of\n                    <jats:italic>Mecp2<\/jats:italic>\n                    knockout mice\u2014even though treatments were initiated only after the mice had already developed symptoms. Stimulation of endogenous cellular pathways may thus be a promising approach for the treatment of RTT patients.\n                  <\/jats:p>","DOI":"10.15252\/emmm.201910889","type":"journal-article","created":{"date-parts":[[2020,1,8]],"date-time":"2020-01-08T09:11:39Z","timestamp":1578474699000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":39,"title":["Huntingtin phosphorylation governs BDNF homeostasis and improves the phenotype of Mecp2 knockout mice"],"prefix":"10.1038","volume":"12","author":[{"given":"Yann","family":"Ehinger","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Julie","family":"Bruy\u00e8re","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicolas","family":"Panayotis","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yah\u2010Se","family":"Abada","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emilie","family":"Borloz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Val\u00e9rie","family":"Matagne","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chiara","family":"Scaramuzzino","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"H\u00e9l\u00e8ne","family":"Vitet","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Benoit","family":"Delatour","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lydia","family":"Saidi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laurent","family":"Villard","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6107-1046","authenticated-orcid":false,"given":"Fr\u00e9d\u00e9ric","family":"Saudou","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9721-8844","authenticated-orcid":false,"given":"Jean\u2010Christophe","family":"Roux","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2020,1,8]]},"reference":[{"key":"BFEMMM201910889_CR1","doi-asserted-by":"crossref","first-page":"856","DOI":"10.1038\/39885","volume":"389","author":"CA Altar","year":"1997","unstructured":"Altar CA, Cai N, Bliven T, Juhasz M, Conner JM, Acheson AL, Lindsay RM, Wiegand SJ (1997) Anterograde transport of brain\u2010derived neurotrophic factor and its role in the brain. 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