{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,28]],"date-time":"2026-10-28T01:57:15Z","timestamp":1793152635915,"version":"4.5.0"},"reference-count":50,"publisher":"Public Library of Science (PLoS)","issue":"3","license":[{"start":{"date-parts":[[2023,3,1]],"date-time":"2023-03-01T00:00:00Z","timestamp":1677628800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"National Center for Advancing Translational Sciences of the National Institutes of Health","award":["UL1TR002378"],"award-info":[{"award-number":["UL1TR002378"]}]},{"DOI":"10.13039\/100006939","name":"Emory University","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100006939","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100016499","name":"Rett Syndrome Research Trust","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100016499","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100016499","name":"Rett Syndrome Research Trust","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100016499","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100016499","name":"Rett Syndrome Research Trust","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100016499","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100016499","name":"Rett Syndrome Research Trust","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100016499","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100006939","name":"Emory University","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100006939","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100006939","name":"Emory University","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100006939","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100006939","name":"Emory University","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100006939","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100016499","name":"Rett Syndrome Research Trust","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100016499","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["www.plosone.org"],"crossmark-restriction":false},"short-container-title":["PLoS ONE"],"abstract":"<jats:p>Rett syndrome, a rare genetic neurodevelopmental disorder in humans, does not have an effective cure. However, multiple therapies and medications exist to treat symptoms and improve patients\u2019 quality of life. As research continues to discover and evaluate new medications for Rett syndrome patients, there remains a lack of objective physiological and motor activity-based (physio-motor) biomarkers that enable the measurement of the effect of these medications on the change in patients\u2019 Rett syndrome severity. In our work, using a commercially available wearable chest patch, we recorded simultaneous electrocardiogram and three-axis acceleration from 20 patients suffering from Rett syndrome along with the corresponding Clinical Global Impression\u2014Severity score, which measures the overall disease severity on a 7-point Likert scale. We derived physio-motor features from these recordings that captured heart rate variability, activity metrics, and the interactions between heart rate and activity. Further, we developed machine learning (ML) models to classify high-severity Rett patients from low-severity Rett patients using the derived physio-motor features. For the best-trained model, we obtained a pooled area under the receiver operating curve equal to 0.92 via a leave-one-out-patient cross-validation approach. Finally, we computed the feature popularity scores for all the trained ML models and identified physio-motor biomarkers for Rett syndrome.<\/jats:p>","DOI":"10.1371\/journal.pone.0266351","type":"journal-article","created":{"date-parts":[[2023,3,1]],"date-time":"2023-03-01T13:24:11Z","timestamp":1677677051000},"page":"e0266351","update-policy":"https:\/\/doi.org\/10.1371\/journal.pone.corrections_policy","source":"Crossref","is-referenced-by-count":10,"title":["Rett syndrome severity estimation with the BioStamp nPoint using interactions between heart rate variability and body movement"],"prefix":"10.1371","volume":"18","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1809-6613","authenticated-orcid":true,"given":"Pradyumna Byappanahalli","family":"Suresha","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Heather","family":"O\u2019Leary","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daniel C.","family":"Tarquinio","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jana","family":"Von Hehn","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gari D.","family":"Clifford","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"340","published-online":{"date-parts":[[2023,3,1]]},"reference":[{"key":"pone.0266351.ref001","first-page":"723","article-title":"On a unusual brain atrophy syndrome in hyperammonemia in childhood","volume":"116","author":"A Rett","year":"1966","journal-title":"Wiener medizinische Wochenschrift (1946)"},{"key":"pone.0266351.ref002","doi-asserted-by":"crossref","first-page":"422","DOI":"10.1016\/j.neuron.2007.10.001","article-title":"The story of Rett syndrome: from clinic to neurobiology","volume":"56","author":"M Chahrour","year":"2007","journal-title":"Neuron"},{"key":"pone.0266351.ref003","doi-asserted-by":"crossref","unstructured":"Tarquinio DC, Percy AK. 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