{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T13:27:18Z","timestamp":1791293238199,"version":"4.1.0"},"reference-count":35,"publisher":"American Physiological Society","issue":"5","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Journal of Applied Physiology"],"published-print":{"date-parts":[[2012,3,1]]},"abstract":"<jats:p>Rett syndrome (RTT), caused by mutations in the methyl-CpG binding protein 2 gene ( MECP2), is a debilitating autism spectrum developmental disorder predominantly affecting females. Mecp2 mutant mice have reduced levels of brain-derived neurotrophic factor (BDNF) in the brain; conditional deletion and overexpression of BDNF in the brain accelerates and slows, respectively, disease progression in Mecp2 mutant mice. Thus we tested the hypothesis that 7,8-dihydroxyflavone (7,8-DHF), a small molecule reported to activate the high affinity BDNF receptor (TrkB) in the CNS, would attenuate disease progression in Mecp2 mutant mice. Following weaning, 7,8-DHF was administered in drinking water throughout life. Treated mutant mice lived significantly longer compared with untreated mutant littermates (80 \u00b1 4 and 66 \u00b1 2 days, respectively). 7,8-DHF delayed body weight loss, increased neuronal nuclei size and enhanced voluntary locomotor (running wheel) distance in Mecp2 mutant mice. In addition, administration of 7,8-DHF partially improved breathing pattern irregularities and returned tidal volumes to near wild-type levels. Thus although the specific mechanisms are not completely known, 7,8-DHF appears to reduce disease symptoms in Mecp2 mutant mice and may have potential as a therapeutic treatment for RTT patients.<\/jats:p>","DOI":"10.1152\/japplphysiol.01361.2011","type":"journal-article","created":{"date-parts":[[2011,12,23]],"date-time":"2011-12-23T05:09:53Z","timestamp":1324616993000},"page":"704-710","source":"Crossref","is-referenced-by-count":99,"title":["7,8-dihydroxyflavone exhibits therapeutic efficacy in a mouse model of Rett syndrome"],"prefix":"10.1152","volume":"112","author":[{"given":"Rebecca A.","family":"Johnson","sequence":"first","affiliation":[{"name":"Department of Surgical Sciences,"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maxine","family":"Lam","sequence":"additional","affiliation":[{"name":"Waisman Center,"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Antonio M.","family":"Punzo","sequence":"additional","affiliation":[{"name":"Department of Surgical Sciences,"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hongda","family":"Li","sequence":"additional","affiliation":[{"name":"Waisman Center,"},{"name":"Laboratory of Genetics,"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Benjamin R.","family":"Lin","sequence":"additional","affiliation":[{"name":"Waisman Center,"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Keqiang","family":"Ye","sequence":"additional","affiliation":[{"name":"Department of Pathology and Laboratory Medicine, Emory University School of Medicine, Atlanta, Georgia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gordon S.","family":"Mitchell","sequence":"additional","affiliation":[{"name":"Department of Comparative Biosciences, and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Qiang","family":"Chang","sequence":"additional","affiliation":[{"name":"Waisman Center,"},{"name":"Laboratory of Genetics,"},{"name":"Department of Neurology, University of Wisconsin-Madison, Madison, Wisconsin; 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