Cargando…

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...

Descripción completa

Detalles Bibliográficos
Autores principales: Wang, Fajun, Darby, Joseph
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2023
Materias:
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
_version_ 1785092066515091456
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
work_keys_str_mv AT wangfajun casereportcentralalveolarhypoventilationinasurvivorofcardiopulmonaryarrest
AT darbyjoseph casereportcentralalveolarhypoventilationinasurvivorofcardiopulmonaryarrest