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Case report: Central alveolar hypoventilation in a survivor of cardiopulmonary arrest
INTRODUCTION: Ondine’s 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 arres...
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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Frontiers Media S.A.
2023
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Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10435288/ https://www.ncbi.nlm.nih.gov/pubmed/37602250 http://dx.doi.org/10.3389/fneur.2023.1195008 |
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author | Wang, Fajun Darby, Joseph |
author_facet | Wang, Fajun Darby, Joseph |
author_sort | Wang, Fajun |
collection | PubMed |
description | INTRODUCTION: Ondine’s 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. CASE PRESENTATION: 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 ng/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 days post arrest. CONCLUSION: 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 weeks. Our case highlighted the essential functions of NTS in regulating the respiratory and cardiovascular systems. |
format | Online Article Text |
id | pubmed-10435288 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | Frontiers Media S.A. |
record_format | MEDLINE/PubMed |
spelling | pubmed-104352882023-08-18 Case report: Central alveolar hypoventilation in a survivor of cardiopulmonary arrest Wang, Fajun Darby, Joseph Front Neurol Neurology INTRODUCTION: Ondine’s 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. CASE PRESENTATION: 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 ng/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 days post arrest. CONCLUSION: 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 weeks. Our case highlighted the essential functions of NTS in regulating the respiratory and cardiovascular systems. Frontiers Media S.A. 2023-08-03 /pmc/articles/PMC10435288/ /pubmed/37602250 http://dx.doi.org/10.3389/fneur.2023.1195008 Text en Copyright © 2023 Wang and Darby. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms. |
spellingShingle | Neurology Wang, Fajun Darby, Joseph Case report: Central alveolar hypoventilation in a survivor of cardiopulmonary arrest |
title | Case report: Central alveolar hypoventilation in a survivor of cardiopulmonary arrest |
title_full | Case report: Central alveolar hypoventilation in a survivor of cardiopulmonary arrest |
title_fullStr | Case report: Central alveolar hypoventilation in a survivor of cardiopulmonary arrest |
title_full_unstemmed | Case report: Central alveolar hypoventilation in a survivor of cardiopulmonary arrest |
title_short | Case report: Central alveolar hypoventilation in a survivor of cardiopulmonary arrest |
title_sort | case report: central alveolar hypoventilation in a survivor of cardiopulmonary arrest |
topic | Neurology |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10435288/ https://www.ncbi.nlm.nih.gov/pubmed/37602250 http://dx.doi.org/10.3389/fneur.2023.1195008 |
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