{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,25]],"date-time":"2026-09-25T07:31:01Z","timestamp":1790321461445,"version":"4.1.0"},"reference-count":64,"publisher":"Society for Neuroscience","issue":"48","license":[{"start":{"date-parts":[[2013,5,28]],"date-time":"2013-05-28T00:00:00Z","timestamp":1369699200000},"content-version":"vor","delay-in-days":181,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-sa\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J. Neurosci."],"published-print":{"date-parts":[[2012,11,28]]},"abstract":"<jats:p>\n                    Rett syndrome (RTT) is a severe neurological disorder that is associated with mutations in the methyl-CpG binding protein 2 (MECP2) gene. RTT patients suffer from mental retardation and behavioral disorders, including heightened anxiety and state-dependent breathing irregularities, such as hyperventilation and apnea. Many symptoms are recapitulated by the\n                    <jats:italic>Mecp2<\/jats:italic>\n                    -null male mice (\n                    <jats:italic>Mecp2<\/jats:italic>\n                    <jats:sup>\n                      \u2212\/\n                      <jats:italic>y<\/jats:italic>\n                    <\/jats:sup>\n                    ). To characterize developmental progression of the respiratory phenotype and explore underlying mechanisms, we examined\n                    <jats:italic>Mecp2<\/jats:italic>\n                    <jats:sup>\n                      \u2212\/\n                      <jats:italic>y<\/jats:italic>\n                    <\/jats:sup>\n                    and wild-type (WT) mice from presymptomatic periods to end-stage disease. We monitored breathing patterns of unrestrained mice during wake\u2013sleep states and while altering stress levels using movement restraint or threatening odorant (trimethylthiazoline). Respiratory motor patterns generated by\n                    <jats:italic>in situ<\/jats:italic>\n                    working heart\u2013brainstem preparations (WHBPs) were measured to assess function of brainstem respiratory networks isolated from suprapontine structures. Data revealed two general stages of respiratory dysfunction in\n                    <jats:italic>Mecp2<\/jats:italic>\n                    <jats:sup>\n                      \u2212\/\n                      <jats:italic>y<\/jats:italic>\n                    <\/jats:sup>\n                    mice. At the early stage, respiratory abnormalities were limited to wakefulness, correlated with markers of stress (increased fecal deposition and blood corticosterone levels), and alleviated by antalarmin (corticotropin releasing hormone receptor 1 antagonist). Furthermore, the respiratory rhythm generated by WHBPs was similar in WT and\n                    <jats:italic>Mecp2<\/jats:italic>\n                    <jats:sup>\n                      \u2212\/\n                      <jats:italic>y<\/jats:italic>\n                    <\/jats:sup>\n                    mice. During the later stage, respiratory abnormalities were evident during wakefulness and sleep. Also, WHBPs from\n                    <jats:italic>Mecp2<\/jats:italic>\n                    <jats:sup>\n                      \u2212\/\n                      <jats:italic>y<\/jats:italic>\n                    <\/jats:sup>\n                    showed central apneas. We conclude that, at early disease stages, stress-related modulation from suprapontine structures is a significant factor in the\n                    <jats:italic>Mecp2<\/jats:italic>\n                    <jats:sup>\n                      \u2212\/\n                      <jats:italic>y<\/jats:italic>\n                    <\/jats:sup>\n                    respiratory phenotype and that anxiolytics may be effective. At later stages, abnormalities of brainstem respiratory networks are a significant cause of irregular breathing patterns and central apneas.\n                  <\/jats:p>","DOI":"10.1523\/jneurosci.2951-12.2012","type":"journal-article","created":{"date-parts":[[2012,11,28]],"date-time":"2012-11-28T12:43:04Z","timestamp":1354106584000},"page":"17230-17240","source":"Crossref","is-referenced-by-count":45,"title":["Anxiety-Related Mechanisms of Respiratory Dysfunction in a Mouse Model of Rett Syndrome"],"prefix":"10.1523","volume":"32","author":[{"given":"Jun","family":"Ren","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiuqing","family":"Ding","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gregory D.","family":"Funk","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"John 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