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翁达恩氏诅咒:膈肌起搏与自主呼吸恢复的临床表现。病例说明。

Ondine's curse: clinical presentation with diaphragmatic pacing and spontaneous respiratory recovery. Illustrative case.

作者信息

Schupper Alexander J, Devarajan Alex, Lee Dong-Seok, Perez Enrique, Shrivastava Raj K

机构信息

Departments of1Neurological Surgery.

2Thoracic Surgery, and.

出版信息

J Neurosurg Case Lessons. 2023 May 22;5(21). doi: 10.3171/CASE233.

DOI:10.3171/CASE233
PMID:37218735
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10550651/
Abstract

BACKGROUND

The complexity of posterior fossa surgery can often lead to rare complications due to the anatomy involved. Vestibular schwannoma resection is a common pathology in the posterior fossa, often requiring surgical intervention. Given the proximity of this space to the brainstem, cranial nerve VII/VIII complex, and posterior inferior cerebellar artery (PICA), neurovascular complications are not infrequent. A rare vascular complication from this surgical approach is a lateral medullary infarction from injury to the lateral medullary segment of the proximal PICA, leading to central hypoventilation syndrome (CHS).

OBSERVATIONS

This report presents a unique case of a 51-year-old man who underwent a retrosigmoid craniectomy for resection of a vestibular schwannoma. Following surgery, the patient was unable to be weaned off the ventilator and was noted to become apneic while he slept, a clinical picture consistent with Ondine's curse.

LESSONS

This report discusses the anatomical considerations of this surgical corridor leading to this complication and the management of a patient with acquired Ondine's curse and reviews the scarce literature on this uncommon cause of acquired CHS.

摘要

背景

由于所涉及的解剖结构,后颅窝手术的复杂性常常会导致罕见的并发症。前庭神经鞘瘤切除术是后颅窝常见的病理情况,通常需要手术干预。鉴于该区域与脑干、颅神经VII/VIII复合体以及小脑后下动脉(PICA)相邻,神经血管并发症并不罕见。这种手术方式罕见的血管并发症是近端PICA外侧延髓段损伤导致的外侧延髓梗死,进而引发中枢性低通气综合征(CHS)。

观察结果

本报告介绍了一例独特病例,一名51岁男性因前庭神经鞘瘤切除术接受了乙状窦后开颅手术。术后,患者无法撤机,且在睡眠时出现呼吸暂停,临床表现符合翁丁氏咒诅综合征。

经验教训

本报告讨论了导致该并发症的此手术通道的解剖学因素以及获得性翁丁氏咒诅综合征患者的管理,并回顾了关于这种罕见的获得性CHS病因的稀少文献。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0e75/10550651/47dc5173ab7f/CASE233f4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0e75/10550651/60791b053018/CASE233f1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0e75/10550651/460fdc8e9c28/CASE233f2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0e75/10550651/4afea354f2f7/CASE233f3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0e75/10550651/47dc5173ab7f/CASE233f4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0e75/10550651/60791b053018/CASE233f1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0e75/10550651/460fdc8e9c28/CASE233f2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0e75/10550651/4afea354f2f7/CASE233f3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0e75/10550651/47dc5173ab7f/CASE233f4.jpg

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本文引用的文献

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Perioperative outcomes and the effects of anesthesia in congenital central hypoventilation patients.先天性中枢性肺泡换气不足患者的围手术期结果和麻醉效果。
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Sleep Apnea Syndrome after Posterior Fossa Surgery: A Case of Acquired Ondine's Curse.后颅窝手术后的睡眠呼吸暂停综合征:一例获得性翁丁氏诅咒病例。
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Extended use of diaphragm pacing in patients with unilateral or bilateral diaphragm dysfunction: a new therapeutic option.膈肌起搏在单侧或双侧膈肌功能障碍患者中的延长应用:一种新的治疗选择。
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Case report of os odontoideum causing Ondine's curse.齿突游离小骨导致翁丁氏诅咒的病例报告。
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Brainstem tethering with Ondine's curse.脑干拴系术与翁丁诅咒。
World Neurosurg. 2011 Dec;76(6):592.e11-4. doi: 10.1016/j.wneu.2011.03.020.
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Paradoxical herniation in wartime penetrating brain injury with concomitant skull-base trauma.战时穿透性脑损伤合并颅底创伤时的反常疝
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Best clinical practices for the sleep center adjustment of noninvasive positive pressure ventilation (NPPV) in stable chronic alveolar hypoventilation syndromes.稳定慢性肺泡低通气综合征患者睡眠中心调整无创正压通气(NPPV)的最佳临床实践。
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