Case report: Central alveolar hypoventilation in a survivor of cardiopulmonary arrest

被引:0
作者
Wang, Fajun [1 ,2 ]
Darby, Joseph [1 ]
机构
[1] Univ Pittsburgh, Dept Crit Care Med, Med Ctr, Pittsburgh, PA 15213 USA
[2] St Louis Univ, Dept Neurol, St Louis, MO 63103 USA
来源
FRONTIERS IN NEUROLOGY | 2023年 / 14卷
关键词
Ondine's curse; central alveolar hypoventilation; cardiac arrest and brain injuries; nucleus tracti solitarii; dysautonomia; RESPIRATORY-FAILURE; SOLITARY TRACT; BRAIN-STEM; NUCLEUS;
D O I
10.3389/fneur.2023.1195008
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
IntroductionOndine'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 presentationA 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. ConclusionWe 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.
引用
收藏
页数:4
相关论文
共 15 条
  • [1] The chemical neuroanatomy of breathing
    Alheid, George F.
    McCrimmon, Donald R.
    [J]. RESPIRATORY PHYSIOLOGY & NEUROBIOLOGY, 2008, 164 (1-2) : 3 - 11
  • [2] RESPIRATORY-FAILURE AND UNILATERAL CAUDAL BRAIN-STEM INFARCTION
    BOGOUSSLAVSKY, J
    KHURANA, R
    DERUAZ, JP
    HORNUNG, JP
    REGLI, F
    JANZER, R
    PERRET, C
    [J]. ANNALS OF NEUROLOGY, 1990, 28 (05) : 668 - 673
  • [3] Adrenergic activation of the nucleus tractus solitarius potentiates amygdala norepinephrine release and enhances retention performance in emotionally arousing and spatial memory tasks
    Clayton, EC
    Williams, CL
    [J]. BEHAVIOURAL BRAIN RESEARCH, 2000, 112 (1-2) : 151 - 158
  • [4] Nucleus of the solitary tract, medullary reflexes, and clinical implications
    Cutsforth-Gregory, Jeremy K.
    Benarroch, Eduardo E.
    [J]. NEUROLOGY, 2017, 88 (12) : 1187 - 1196
  • [5] De Caro R, 2000, STROKE, V31, P1187
  • [6] Sleep-related disorders and their relationship with MRI findings in multiple sclerosis
    Foschi, M.
    Rizzo, G.
    Liguori, R.
    Avoni, P.
    Mancinelli, L.
    Lugaresi, A.
    Ferini-Strambi, L.
    [J]. SLEEP MEDICINE, 2019, 56 : 90 - 97
  • [7] Chemosensitive Phox2b-expressing neurons are crucial for hypercapnic ventilatory response in the nucleus tractus solitarius
    Fu, Congrui
    Xue, Jinyu
    Wang, Ri
    Chen, Jinting
    Ma, Lan
    Liu, Yixian
    Wang, Xuejiao
    Guo, Fang
    Zhang, Yi
    Zhang, Xiangjian
    Wang, Sheng
    [J]. JOURNAL OF PHYSIOLOGY-LONDON, 2017, 595 (14): : 4973 - 4989
  • [8] Acute Respiratory Failure due to Unilateral Dorsolateral Bulbar Infarction
    Kumral, Emre
    Uzunkopru, Cihat
    Ciftci, Seyma
    Demirci, Tenzile
    [J]. EUROPEAN NEUROLOGY, 2011, 66 (02) : 70 - 74
  • [9] THE LOCATION AND FUNCTION OF RESPIRATORY FIBERS IN THE 2ND CERVICAL SPINAL-CORD SEGMENT - RESPIRATORY DYSFUNCTION SYNDROME AFTER CERVICAL CORDOTOMY
    LAHUERTA, J
    BUXTON, P
    LIPTON, S
    BOWSHER, D
    [J]. JOURNAL OF NEUROLOGY NEUROSURGERY AND PSYCHIATRY, 1992, 55 (12) : 1142 - 1145
  • [10] Progression rates of medulla oblongata infarcts in clinical follow-up: A retrospective study
    Memmedova, Fergane
    Sevingil, Semra Ari
    Jafarova, Ulviyyat
    Cifter, Gizem
    Aykac, Ozlem
    Ozdemir, Atilla Ozcan
    [J]. CLINICAL NEUROLOGY AND NEUROSURGERY, 2022, 219