{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,8]],"date-time":"2026-07-08T19:09:02Z","timestamp":1783537742281,"version":"3.55.0"},"reference-count":33,"publisher":"Public Library of Science (PLoS)","issue":"7","license":[{"start":{"date-parts":[[2026,7,8]],"date-time":"2026-07-08T00:00:00Z","timestamp":1783468800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100001665","name":"Agence Nationale de la Recherche","doi-asserted-by":"publisher","award":["Investissements d\u2019avenir \u00bb Program Grants ANR-10-IAIHU-06"],"award-info":[{"award-number":["Investissements d\u2019avenir \u00bb Program Grants ANR-10-IAIHU-06"]}],"id":[{"id":"10.13039\/501100001665","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Fondation Assistance Publique-H\u00f4pitaux de Paris","award":["EPIRES \u2013 Marie Laure PLV Merchandising"],"award-info":[{"award-number":["EPIRES \u2013 Marie Laure PLV Merchandising"]}]},{"DOI":"10.13039\/501100002915","name":"Fondation pour la Recherche M\u00e9dicale","doi-asserted-by":"publisher","award":["FDM202106013478"],"award-info":[{"award-number":["FDM202106013478"]}],"id":[{"id":"10.13039\/501100002915","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["www.plosone.org"],"crossmark-restriction":false},"short-container-title":["PLoS One"],"abstract":"<jats:p>Lance-Adams syndrome is a severe and disabling posthypoxic myoclonus in humans. Its underlying mechanisms are still unknown. We aimed to develop a new animal model of Lance-Adams syndrome. We performed our procedures on Sprague-Dawley rats monitored for basic cardiorespiratory parameters. The Group 1 was sedated and submitted to hypoxia in a dedicated cage where oxygen was replaced with nitrogen. The Groups 2 and 3 were sedated, intubated, ventilated, and submitted to anoxia with the replacement of oxygen by nitrogen in the ventilatory circuit, either continuously (Group 2) or intermittently (Group 3). Rats benefited from cardiopulmonary resuscitation. Each rat was evaluated daily for spontaneous, wandering-induced, and auditory stimuli-induced myoclonic jerks, for the latter using a quantitative myoclonus score. In the Group 1 (n\u2009=\u200919), the duration of hypoxia was 25\u2009\u00b1\u200920 min for the surviving animals (n\u2009=\u200914\/19) and minimal partial pressure of oxygen in the cage was 7.2\u2009\u00b1\u20090.8%. In the Group 2 (n\u2009=\u200938), the duration of anoxia was 5.6\u2009\u00b1\u20091.9 min for the surviving animals (n\u2009=\u200923\/38). In the Group 3 (n\u2009=\u200930), the total duration of anoxia was 15\u2009\u00b1\u20093.5 min in the surviving animals (n\u2009=\u200915\/30). These three procedures did not allow for generating a myoclonic phenotype. Some refinements of hypoxic\/anoxic procedures are still needed to develop an animal model of posthypoxic myoclonus that would be a precious tool to understand better the pathophysiology of Lance-Adams syndrome.<\/jats:p>","DOI":"10.1371\/journal.pone.0317638","type":"journal-article","created":{"date-parts":[[2026,7,8]],"date-time":"2026-07-08T17:46:36Z","timestamp":1783532796000},"page":"e0317638","update-policy":"https:\/\/doi.org\/10.1371\/journal.pone.corrections_policy","source":"Crossref","is-referenced-by-count":0,"title":["Towards new animal models of pure hypoxic Lance-Adams syndrome: Negative results"],"prefix":"10.1371","volume":"21","author":[{"given":"Geoffroy","family":"Vellieux","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Delphine","family":"Roussel","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anthony","family":"Pinto","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nathalie","family":"Mougenot","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0077-8114","authenticated-orcid":true,"given":"Vincent","family":"Navarro","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"340","published-online":{"date-parts":[[2026,7,8]]},"reference":[{"key":"pone.0317638.ref001","doi-asserted-by":"crossref","first-page":"111","DOI":"10.1093\/brain\/86.1.111","article-title":"The syndrome of intention or action myoclonus as a sequel to hypoxic encephalopathy","volume":"86","author":"JW LANCE","year":"1963","journal-title":"Brain"},{"issue":"5","key":"pone.0317638.ref002","doi-asserted-by":"crossref","DOI":"10.1093\/braincomms\/fcaf329","article-title":"Lance-Adams syndrome or chronic post-hypoxic myoclonus in adults: a systematic literature review","volume":"7","author":"G Vellieux","year":"2025","journal-title":"Brain Commun"},{"key":"pone.0317638.ref003","doi-asserted-by":"crossref","first-page":"98","DOI":"10.1016\/j.cnp.2017.03.003","article-title":"Post-hypoxic myoclonus: Differentiating benign and malignant etiologies in diagnosis and prognosis","volume":"2","author":"B Freund","year":"2017","journal-title":"Clin Neurophysiol Pract"},{"issue":"6","key":"pone.0317638.ref004","doi-asserted-by":"crossref","first-page":"399","DOI":"10.1159\/000509318","article-title":"Clinical Outcome and Intraoperative Neurophysiology of the Lance-Adams Syndrome Treated with Bilateral Deep Brain Stimulation of the Globus Pallidus Internus: A Case Report and Review of the Literature","volume":"98","author":"H Mure","year":"2020","journal-title":"Stereotact Funct Neurosurg"},{"issue":"2","key":"pone.0317638.ref005","doi-asserted-by":"crossref","first-page":"647","DOI":"10.1016\/j.brs.2023.03.004","article-title":"Effectiveness of electroconvulsive therapy in Lance-Adams syndrome","volume":"16","author":"G Vellieux","year":"2023","journal-title":"Brain Stimul"},{"issue":"11","key":"pone.0317638.ref006","doi-asserted-by":"crossref","DOI":"10.1212\/WNL.0000000000209994","article-title":"Multimodal Assessment of the Origin of Myoclonus in Lance-Adams Syndrome","volume":"103","author":"G Vellieux","year":"2024","journal-title":"Neurology"},{"issue":"2","key":"pone.0317638.ref007","doi-asserted-by":"crossref","first-page":"201","DOI":"10.1002\/mds.870090214","article-title":"Novel rat cardiac arrest model of posthypoxic myoclonus","volume":"9","author":"DD Truong","year":"1994","journal-title":"Mov Disord"},{"issue":"2","key":"pone.0317638.ref008","doi-asserted-by":"crossref","first-page":"238","DOI":"10.1038\/jcbfm.1992.34","article-title":"Global cerebral ischemia associated with cardiac arrest in the rat: I. 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Courten-Myers","year":"1985","journal-title":"Stroke"},{"key":"pone.0317638.ref020","doi-asserted-by":"crossref","first-page":"72","DOI":"10.1016\/j.neuroscience.2019.05.042","article-title":"Oxygen-Glucose Deprivation Differentially Affects Neocortical Pyramidal Neurons and Parvalbumin-Positive Interneurons","volume":"412","author":"N Povysheva","year":"2019","journal-title":"Neuroscience"},{"key":"pone.0317638.ref021","doi-asserted-by":"crossref","first-page":"1087725","DOI":"10.3389\/fnins.2022.1087725","article-title":"Rat model of asphyxia-induced cardiac arrest and resuscitation","volume":"16","author":"S Yu","year":"2023","journal-title":"Front Neurosci"},{"issue":"11","key":"pone.0317638.ref022","doi-asserted-by":"crossref","DOI":"10.1093\/nsr\/nwad248","article-title":"Neurogenesis in primates versus rodents and the value of non-human primate models","volume":"10","author":"R Zhang","year":"2023","journal-title":"Natl Sci 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Sci"},{"issue":"5","key":"pone.0317638.ref029","doi-asserted-by":"crossref","first-page":"834","DOI":"10.1248\/bpb.b14-00012","article-title":"Inhalation anesthesia is preferable for recording rat cardiac function using an electrocardiogram","volume":"37","author":"M Murakami","year":"2014","journal-title":"Biol Pharm Bull"},{"issue":"8","key":"pone.0317638.ref030","doi-asserted-by":"crossref","first-page":"1679","DOI":"10.1161\/01.STR.29.8.1679","article-title":"Normoxic ventilation after cardiac arrest reduces oxidation of brain lipids and improves neurological outcome","volume":"29","author":"Y Liu","year":"1998","journal-title":"Stroke"},{"issue":"1","key":"pone.0317638.ref031","doi-asserted-by":"crossref","first-page":"3419","DOI":"10.1038\/s41598-023-30120-1","article-title":"Bio-physiological susceptibility of the brain, heart, and lungs to systemic ischemia reperfusion and hyperoxia-induced injury in post-cardiac arrest rats","volume":"13","author":"T 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