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脑干离断:另外两名患者及表型扩展

Brainstem disconnection: two additional patients and expansion of the phenotype.

作者信息

Poretti Andrea, Denecke Jonas, Miller Douglas C, Schiffmann Holger, Buhk Jan Hendrik, Grange Dorothy K, Doherty Dan, Boltshauser Eugen

机构信息

Section of Pediatric Neuroradiology, Division of Pediatric Radiology, Russell H. Morgan Department of Radiology and Radiological Science, The Johns Hopkins University School of Medicine, Baltimore, Maryland, United States.

Department of Pediatrics, University Medical Center Hamburg-Eppendorf, Hamburg, Germany.

出版信息

Neuropediatrics. 2015 Apr;46(2):139-44. doi: 10.1055/s-0034-1544127. Epub 2015 Feb 11.

Abstract

Brainstem disconnection (BD) is a rare posterior fossa abnormality defined by the nearly complete absence of a brainstem segment with the rostral and caudal brainstem portions connected only by a thin cord of tissue. The outcome is poor and the majority of children die within the first 2 months of life without achieving developmental milestones. We report on the cases of two children with BD and a prolonged spontaneous survival. Neither patient required intubation or mechanical ventilation and each survived longer than 2 months (one child died at the age of 8 months, the other is alive at the age of 4.5 years). In addition, patient 1 is the only child with BD reported so far who achieved some developmental milestones. Although the long-term neurodevelopmental outcome of BD remains unfavorable, the expansion of the phenotypic spectrum may be important in terms of counseling.

摘要

脑干离断(BD)是一种罕见的后颅窝异常,其定义为脑干节段几乎完全缺失,脑干的头端和尾端部分仅通过一条细索状组织相连。预后很差,大多数患儿在出生后的头2个月内死亡,未达到发育里程碑。我们报告了2例脑干离断患儿长期自然存活的病例。两名患儿均无需插管或机械通气,且均存活超过2个月(一名患儿8个月时死亡,另一名患儿4.5岁时仍存活)。此外,患者1是迄今为止报道的唯一一名达到一些发育里程碑的脑干离断患儿。尽管脑干离断的长期神经发育预后仍然不佳,但在咨询方面,扩大表型谱可能很重要。

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