Byard R W
Department of Paediatrics, University of Adelaide, Australia.
Am J Forensic Med Pathol. 1996 Sep;17(3):260-3. doi: 10.1097/00000433-199609000-00015.
Neurological and vascular complications of Arnold-Chiari malformation treated with ventriculoatrial shunting may result in sudden or unexpected death. Two patients with Arnold-Chiari malformation and ventriculoatrial shunting had variable clinical manifestations and diagnostic difficulties. A 3-year-old girl with a 1-day history of right-sided heart failure died unexpectedly soon after cardiac catheterization. At autopsy examination an adherent thrombus around the ventriculoatrial catheter tip, pulmonary infarction, and embolic pulmonary arterial hypertensive changes were found. In the second case, a 21-year-old man died suddenly after a brief episode of dyspnea. He had a 1-year history of "asthma" before death. Autopsy examination confirmed pulmonary infarction and embolic pulmonary arterial hypertensive changes. There was no histological evidence of asthma. Deaths in both cases were due to pulmonary infarction stemming from thromboemboli derived from ventriculoatrial catheterization. Both patients had evidence of long-standing clinically unsuspected vascular disease, which may have contributed to death. Cardiac catheterization may also have precipitated death in the first patient. Other possible problems leading or contributing to sudden death in such patients include pulmonary hypertension with chronic cor pulmonale, airway obstruction from recurrent laryngeal nerve paralysis, and shunt blockage with acute hydrocephalus. Lethal brainstem compression may also accompany relatively minor trauma associated with chronic cerebellar tonsillar herniation in these patients.
采用脑室心房分流术治疗阿诺德-奇阿利畸形时,神经和血管并发症可能导致猝死或意外死亡。两名患有阿诺德-奇阿利畸形并接受脑室心房分流术的患者临床表现各异,诊断存在困难。一名3岁女童有1天的右侧心力衰竭病史,在心脏导管插入术后不久意外死亡。尸检发现脑室心房导管尖端周围有粘连性血栓、肺梗死和栓塞性肺动脉高压改变。在第二例中,一名21岁男性在短暂的呼吸困难发作后突然死亡。他在死前有1年的“哮喘”病史。尸检证实有肺梗死和栓塞性肺动脉高压改变。没有哮喘的组织学证据。两例死亡均归因于脑室心房导管插入术引起的血栓栓塞导致的肺梗死。两名患者均有长期临床未被怀疑的血管疾病证据,这可能是导致死亡的原因。心脏导管插入术也可能是第一例患者死亡的诱因。此类患者导致或促成猝死的其他可能问题包括慢性肺源性心脏病伴有的肺动脉高压、喉返神经麻痹引起的气道阻塞以及分流堵塞伴急性脑积水。在这些患者中,与慢性小脑扁桃体疝相关的相对轻微创伤也可能伴有致命的脑干压迫。