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目睹的婴儿期睡眠相关性癫痫发作及婴儿猝死综合征:一例报告

Witnessed sleep-related seizure and sudden unexpected death in infancy: a case report.

作者信息

Kinney Hannah C, McDonald Anna G, Minter Megan E, Berry Gerard T, Poduri Annapurna, Goldstein Richard D

机构信息

Department of Pathology, Boston Children's Hospital and Harvard Medical School, Boston, MA 02115, USA.

出版信息

Forensic Sci Med Pathol. 2013 Sep;9(3):418-21. doi: 10.1007/s12024-013-9448-0. Epub 2013 Jul 14.

Abstract

Witnessed reports of sudden death are rare, but critical to deciphering its mechanism(s). We report such a death in a seemingly healthy 8-month-old boy in whom seizures and respiratory distress in the prone position were witnessed upon discovery during a sleep period. Following cardiopulmonary resuscitation, anoxic encephalopathy resulted in "brain death" and withdrawal of life support after 2 days. The autopsy did not reveal a primary anatomic cause of death. Metabolic evaluation failed to uncover an inborn error of ammonia, amino, organic, or fatty acid metabolism. Seizures in this case may have been secondary to cerebral hypoxia-ischemia complicating cardiorespiratory arrest of unknown etiology. Yet, they may represent the first manifestation of idiopathic epilepsy, triggering cardiopulmonary arrest, analogous to the terminal events postulated in sudden and unexplained death in epilepsy. This report alerts the forensic community to the possibility that sudden and unexplained death in infants may be due to seizures.

摘要

目击到的猝死报告很少见,但对于解读其机制至关重要。我们报告了一名看似健康的8个月大男婴的死亡案例,在睡眠期间被发现时,他处于俯卧位,伴有癫痫发作和呼吸窘迫。经过心肺复苏,缺氧性脑病导致“脑死亡”,两天后停止了生命支持。尸检未发现主要的解剖学死因。代谢评估未能发现氨、氨基酸、有机或脂肪酸代谢的先天性缺陷。该病例中的癫痫发作可能继发于病因不明的心肺骤停并发的脑缺氧缺血。然而,它们可能代表特发性癫痫的首发表现,引发心肺骤停,类似于癫痫猝死中假定的终末事件。本报告提醒法医界注意婴儿猝死且死因不明可能是由癫痫发作所致的可能性。

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本文引用的文献

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Hippocampal asymmetry and sudden unexpected death in infancy: a case report.海马体不对称与婴儿期意外猝死:一例报告
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