{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,1]],"date-time":"2026-02-01T20:48:18Z","timestamp":1769978898280,"version":"3.49.0"},"reference-count":15,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2023,8,3]],"date-time":"2023-08-03T00:00:00Z","timestamp":1691020800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Neurol."],"abstract":"<jats:sec><jats:title>Introduction<\/jats:title><jats:p>Ondine\u2019s curse is a rare respiratory disorder that is characterized by central alveolar hypoventilation (CAH) during sleep. It is most commonly congenital. However, it can also be acquired very rarely. Herein, we report a young survivor who developed CAH following cardiopulmonary arrest.<\/jats:p><\/jats:sec><jats:sec><jats:title>Case presentation<\/jats:title><jats:p>A 35-year-old man was admitted to the Intensive Care Unit following unwitnessed cardiopulmonary arrest. Following resuscitative interventions, he remained comatose. Early diagnostic testing showed elevated neuronal specific enolase (28.7\u2009ng\/ml), absent cortical responses on evoked potential testing and MRI evidence of restricted diffusion in the cerebellum, hippocampi, juxtacortical white matter, superior cerebellar peduncles, dorsal pons, dorsolateral medulla, and upper cervical spinal cord. Ten days following admission, the patient remained comatose and underwent tracheostomy. He subsequently began to emerge from coma but had persistent unexplained hypotension and bradypnea necessitating ongoing vasopressor and respiratory support. Repeat MRI on hospital day 40 revealed residual FLAIR hyperintensities in the medulla within the nucleus tractus solitarius (NTS). After being discharged to long-term acute care facility, he was successfully liberated from mechanical ventilation 70\u2009days post arrest.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>We report the first survivor of cardiopulmonary arrest who was complicated by CAH and hypotension with MRI verified ischemic injury to the bilateral NTS regions. Despite this injury, ventilator and vasopressor dependency resolved over a period of 10\u2009weeks. Our case highlighted the essential functions of NTS in regulating the respiratory and cardiovascular systems.<\/jats:p><\/jats:sec>","DOI":"10.3389\/fneur.2023.1195008","type":"journal-article","created":{"date-parts":[[2023,8,3]],"date-time":"2023-08-03T14:56:45Z","timestamp":1691074605000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":3,"title":["Case report: Central alveolar hypoventilation in a survivor of cardiopulmonary arrest"],"prefix":"10.3389","volume":"14","author":[{"given":"Fajun","family":"Wang","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Joseph","family":"Darby","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2023,8,3]]},"reference":[{"key":"ref1","doi-asserted-by":"publisher","first-page":"70","DOI":"10.1159\/000327538","article-title":"Acute respiratory failure due to unilateral dorsolateral bulbar infarction","volume":"66","author":"Kumral","year":"2011","journal-title":"Eur Neurol"},{"key":"ref2","doi-asserted-by":"publisher","first-page":"668","DOI":"10.1002\/ana.410280511","article-title":"Respiratory failure and unilateral caudal brainstem infarction","volume":"28","author":"Bogousslavsky","year":"1990","journal-title":"Ann Neurol"},{"key":"ref3","doi-asserted-by":"publisher","first-page":"90","DOI":"10.1016\/j.sleep.2019.01.010","article-title":"Sleep-related disorders and their relationship with MRI findings in multiple sclerosis","volume":"56","author":"Foschi","year":"2019","journal-title":"Sleep Med"},{"key":"ref4","doi-asserted-by":"publisher","first-page":"107329","DOI":"10.1016\/j.clineuro.2022.107329","article-title":"Progression rates of medulla oblongata infarcts in clinical follow-up: a retrospective study","volume":"219","author":"Memmedova","year":"2022","journal-title":"Clin Neurol Neurosurg"},{"key":"ref5","doi-asserted-by":"publisher","first-page":"e13","DOI":"10.1212\/WNL.0b013e31827b9096","article-title":"Pearls and oy-sters: reversible Ondine\u2019s curse in a case of lateral medullary infarction","volume":"80","author":"Mendoza","year":"2013","journal-title":"Neurology"},{"key":"ref6","doi-asserted-by":"publisher","first-page":"zsx044","DOI":"10.1093\/sleep\/zsx044","article-title":"Sleep-wake disturbances after traumatic brain injury: synthesis of human and animal studies","volume":"40","author":"Sandsmark","year":"2017","journal-title":"Sleep"},{"key":"ref7","doi-asserted-by":"publisher","first-page":"438","DOI":"10.1016\/j.chest.2018.10.002","article-title":"Sleep-disordered breathing and spinal cord injury: a state-of-the-art review","volume":"155","author":"Sankari","year":"2019","journal-title":"Chest"},{"key":"ref8","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1155\/2018\/4894820","article-title":"Central hypoventilation: a rare complication of Wallenberg syndrome","volume":"2018","author":"Sivakumar","year":"2018","journal-title":"Case Rep Neurol Med"},{"key":"ref9","doi-asserted-by":"publisher","first-page":"1187","DOI":"10.1212\/WNL.0000000000003751","article-title":"Nucleus of the solitary tract, medullary reflexes, and clinical implications","volume":"88","author":"Cutsforth-Gregory","year":"2017","journal-title":"Neurology"},{"key":"ref10","doi-asserted-by":"publisher","first-page":"151","DOI":"10.1016\/S0166-4328(00)00178-9","article-title":"Adrenergic activation of the nucleus tractus solitarius potentiates amygdala norepinephrine release and enhances retention performance in emotionally arousing and spatial memory tasks","volume":"112","author":"Clayton","year":"2000","journal-title":"Behav Brain Res"},{"key":"ref11","doi-asserted-by":"publisher","first-page":"3","DOI":"10.1016\/j.resp.2008.07.014","article-title":"The chemical neuroanatomy of breathing","volume":"164","author":"Alheid","year":"2008","journal-title":"Respir Physiol Neurobiol"},{"key":"ref12","doi-asserted-by":"publisher","first-page":"1142","DOI":"10.1136\/jnnp.55.12.1142","article-title":"The location and function of respiratory fibres in the second cervical spinal cord segment: respiratory dysfunction syndrome after cervical cordotomy","volume":"55","author":"Lahuerta","year":"1992","journal-title":"J Neurol Neurosurg Psychiatry"},{"key":"ref13","doi-asserted-by":"publisher","first-page":"71","DOI":"10.1016\/j.ejpn.2003.12.005","article-title":"Watershed infarcts in the fetal and neonatal brainstem. An aetiology of central hypoventilation, dysphagia, Moibius syndrome and micrognathia","volume":"8","author":"Sarnat","year":"2004","journal-title":"Eur J Paediatr Neurol"},{"key":"ref14","doi-asserted-by":"publisher","first-page":"1187","DOI":"10.1161\/01.STR.31.5.1187","article-title":"Solitary tract nuclei in acute heart failure","volume":"31","author":"De Caro","year":"2000","journal-title":"Stroke"},{"key":"ref15","doi-asserted-by":"publisher","first-page":"4973","DOI":"10.1113\/JP274437","article-title":"Chemosensitive Phox2b-expressing neurons are crucial for hypercapnic ventilatory response in the nucleus tractus solitarius","volume":"595","author":"Fu","year":"2017","journal-title":"J Physiol"}],"container-title":["Frontiers in Neurology"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fneur.2023.1195008\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,3]],"date-time":"2023-08-03T14:56:49Z","timestamp":1691074609000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fneur.2023.1195008\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,8,3]]},"references-count":15,"alternative-id":["10.3389\/fneur.2023.1195008"],"URL":"https:\/\/doi.org\/10.3389\/fneur.2023.1195008","relation":{},"ISSN":["1664-2295"],"issn-type":[{"value":"1664-2295","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,8,3]]},"article-number":"1195008"}}