{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2023,9,15]],"date-time":"2023-09-15T05:10:03Z","timestamp":1694754603984},"reference-count":29,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2013,12,19]],"date-time":"2013-12-19T00:00:00Z","timestamp":1387411200000},"content-version":"vor","delay-in-days":18,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["CP Mouse Biology"],"published-print":{"date-parts":[[2013,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Mouse models recapitulate many symptoms of Rett Syndrome, an X\u2010linked disorder caused by mutations in methyl\u2010CpG\u2010binding protein 2 (<jats:italic>MECP2<\/jats:italic>). The study of <jats:italic>Mecp2<\/jats:italic>\u2010null male mice has provided insight into pathogenesis of the disorder\u2014most recently, dysregulation of cholesterol and lipid metabolism. Perisymptomatic treatment with statin drugs successfully mitigates the effects of this metabolic syndrome, increases longevity, and improves motor function. Described here is a metabolic drug screening protocol and timeline for symptom evaluation in <jats:italic>Mecp2<\/jats:italic>\u2010mutant mice. Specifically, mice are treated twice weekly with a compound of interest alongside subjective health assessments, bi\u2010weekly body composition measurements, and blood chemistries. Throughout treatment, behavioral phenotyping tests are carried out at specific time points. This protocol is highly adaptable to other neurological diseases; however, the time for completion depends on the specific mutant model under study. The protocol highlights the use of techniques described in several different <jats:italic>Current Protocols in Mouse Biology<\/jats:italic> articles to carry out testing in a preclinical model. <jats:italic>Curr. Protoc. Mouse Biol<\/jats:italic>. 3:187\u2010204 \u00a9 2013 by John Wiley &amp; Sons, Inc.<\/jats:p>","DOI":"10.1002\/9780470942390.mo130157","type":"journal-article","created":{"date-parts":[[2013,12,19]],"date-time":"2013-12-19T20:27:12Z","timestamp":1387484832000},"page":"187-204","source":"Crossref","is-referenced-by-count":3,"title":["Evaluation of Rett Syndrome Symptom Improvement by Metabolic Modulators in <i>Mecp2<\/i>\u2010Mutant Mice"],"prefix":"10.1002","volume":"3","author":[{"given":"Christie M.","family":"Buchovecky","sequence":"first","affiliation":[{"name":"Department of Molecular and Human Genetics, Baylor College of Medicine  Houston Texas"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Misty G.","family":"Hill","sequence":"additional","affiliation":[{"name":"Department of Molecular and Human Genetics, Baylor College of Medicine  Houston Texas"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jennifer M.","family":"Borkey","sequence":"additional","affiliation":[{"name":"Department of Molecular and Human Genetics, Baylor College of Medicine  Houston Texas"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Stephanie M.","family":"Kyle","sequence":"additional","affiliation":[{"name":"Department of Molecular and Human Genetics, Baylor College of Medicine  Houston Texas"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Monica J.","family":"Justice","sequence":"additional","affiliation":[{"name":"Department of Molecular and Human Genetics, Baylor College of Medicine  Houston Texas"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2013,12,19]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/9780470942390.mo100118"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11940-011-0142-9"},{"key":"e_1_2_8_4_1","doi-asserted-by":"crossref","first-page":"37","DOI":"10.1002\/9780470942390.mo110165","article-title":"Assessment of motor coordination and balance in mice using the rotarod, elevated bridge, and footprint tests","volume":"2","author":"Brooks S.P.","year":"2011","journal-title":"Curr. Protoc. Mouse Biol."},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1002\/9780470942390.mo110118"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.2714"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jacl.2011.06.007"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/0471142735.im0107s73"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1002\/0471142735.im0108s73"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1152\/ajpgi.1999.276.5.G1117"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1242\/dmm.011007"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.resp.2008.04.005"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2012.01.034"},{"key":"e_1_2_8_14_1","doi-asserted-by":"crossref","first-page":"25","DOI":"10.1002\/9780470942390.mo110132","article-title":"Acoustic startle reflex and prepulse inhibition","volume":"2","author":"Ouagazzal A.M.","year":"2011","journal-title":"Curr. Protoc. Mouse Biol."},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.plipres.2011.06.002"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddt111"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.resp.2013.06.022"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1002\/9780470942390.mo130054"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1002\/9780470942390.mo130043"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/dds406"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.febslet.2013.05.042"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1471-4159.2011.07351.x"},{"key":"e_1_2_8_23_1","first-page":"32","article-title":"Anti hyperlipidemic activity of Spirulina platensis in Triton X\u2010100 induced hyperlipidemic rats","volume":"3","author":"Sudha S.S.","year":"2011","journal-title":"Hygeria JD Med."},{"key":"e_1_2_8_24_1","doi-asserted-by":"publisher","DOI":"10.1002\/9780470942390.mo110001"},{"key":"e_1_2_8_25_1","doi-asserted-by":"publisher","DOI":"10.1002\/9780470942390.mo100170"},{"key":"e_1_2_8_26_1","doi-asserted-by":"publisher","DOI":"10.1002\/hep.510280425"},{"key":"e_1_2_8_27_1","first-page":"614","article-title":"Administration of substances to laboratory animals: Equipment considerations, vehicle selection, and solute preparation","volume":"50","author":"Turner P.V.","year":"2011","journal-title":"J. Am. Assoc. Lab. Anim. Sci."},{"key":"e_1_2_8_28_1","first-page":"600","article-title":"Administration of substances to laboratory animals: Routes of administration and factors to consider","volume":"50","author":"Turner P.V.","year":"2011","journal-title":"J. Am. Assoc. Lab. Anim. Sci."},{"key":"e_1_2_8_29_1","doi-asserted-by":"publisher","DOI":"10.1242\/dmm.010124"},{"key":"e_1_2_8_30_1","volume-title":"Mouse Behavioral Testing: How to Use Mice in Behavioral Neuroscience","author":"Wahlsten D.","year":"2010"}],"container-title":["Current Protocols in Mouse Biology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2F9780470942390.mo130157","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/currentprotocols.onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/9780470942390.mo130157","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,14]],"date-time":"2023-09-14T17:58:31Z","timestamp":1694714311000},"score":1,"resource":{"primary":{"URL":"https:\/\/currentprotocols.onlinelibrary.wiley.com\/doi\/10.1002\/9780470942390.mo130157"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2013,12]]},"references-count":29,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2013,12]]}},"alternative-id":["10.1002\/9780470942390.mo130157"],"URL":"https:\/\/doi.org\/10.1002\/9780470942390.mo130157","archive":["Portico"],"relation":{},"ISSN":["2161-2617","2161-2617"],"issn-type":[{"value":"2161-2617","type":"print"},{"value":"2161-2617","type":"electronic"}],"subject":[],"published":{"date-parts":[[2013,12]]}}}