{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T13:28:21Z","timestamp":1791293301698,"version":"4.1.0"},"reference-count":141,"publisher":"The Company of Biologists","issue":"6","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2012,11,1]]},"abstract":"<jats:p>In September of 2011, the National Institute of Neurological Disorders and Stroke (NINDS), the Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), the International Rett Syndrome Foundation (IRSF) and the Rett Syndrome Research Trust (RSRT) convened a workshop involving a broad cross-section of basic scientists, clinicians and representatives from the National Institutes of Health (NIH), the US Food and Drug Administration (FDA), the pharmaceutical industry and private foundations to assess the state of the art in animal studies of Rett syndrome (RTT). The aim of the workshop was to identify crucial knowledge gaps and to suggest scientific priorities and best practices for the use of animal models in preclinical evaluation of potential new RTT therapeutics. This review summarizes outcomes from the workshop and extensive follow-up discussions among participants, and includes: (1) a comprehensive summary of the physiological and behavioral phenotypes of RTT mouse models to date, and areas in which further phenotypic analyses are required to enhance the utility of these models for translational studies; (2) discussion of the impact of genetic differences among mouse models, and methodological differences among laboratories, on the expression and analysis, respectively, of phenotypic traits; and (3) definitions of the standards that the community of RTT researchers can implement for rigorous preclinical study design and transparent reporting to ensure that decisions to initiate costly clinical trials are grounded in reliable preclinical data.<\/jats:p>","DOI":"10.1242\/dmm.011007","type":"journal-article","created":{"date-parts":[[2012,10,31]],"date-time":"2012-10-31T13:33:41Z","timestamp":1351690421000},"page":"733-745","source":"Crossref","is-referenced-by-count":195,"title":["Preclinical research in Rett syndrome: setting the foundation for translational success"],"prefix":"10.1242","volume":"5","author":[{"given":"David M.","family":"Katz","sequence":"first","affiliation":[{"name":"Department of Neurosciences, Case Western Reserve University School of Medicine, 10900 Euclid Avenue, Cleveland, OH 44120, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joanne E.","family":"Berger-Sweeney","sequence":"additional","affiliation":[{"name":"Department of Biology, Tufts University, 165 Packard Avenue, Medford, MA 02155, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James H.","family":"Eubanks","sequence":"additional","affiliation":[{"name":"Toronto Western Research Institute, 399 Bathurst Street, Mac 11-408, Toronto, ON M5T 2S8, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Monica J.","family":"Justice","sequence":"additional","affiliation":[{"name":"Department of Molecular and Human Genetics, Baylor College of Medicine, One Baylor Plaza, MS227, R804, Houston, TX 77030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeffrey L.","family":"Neul","sequence":"additional","affiliation":[{"name":"Department of Molecular and Human Genetics, Baylor College of Medicine, One Baylor Plaza, MS227, R804, Houston, TX 77030, USA"},{"name":"Departments of Pediatrics, Neurology, and Neuroscience, Baylor College of Medicine, and Jan and Dan Duncan Neurological Research Institute, Texas Children\u2019s Hospital, 1250 Moursund Street, Houston, TX 77030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lucas","family":"Pozzo-Miller","sequence":"additional","affiliation":[{"name":"Department of Neurobiology, The University of Alabama at Birmingham, 1825 University Boulevard, SHEL-1002, Birmingham, AL 35294, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mary E.","family":"Blue","sequence":"additional","affiliation":[{"name":"Hugo W. 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