Department of Neurology, Universidade de São Paulo, São Paulo, Brazil.
J Neurol Sci. 2011 Jan 15;300(1-2):155-6. doi: 10.1016/j.jns.2010.10.010. Epub 2010 Nov 5.
Brown-Vialetto-Van Laere syndrome (BVVLS) is a rare neurological disease characterized by sensorineural hearing loss and multiple cranial nerve palsies, usually involving the VIIth and IXth to XIIth cranial nerves. We describe the clinical and pathological features of a 33-year-old woman with BVVLS. The patient developed progressive exertional dyspnea, with clinical and laboratory findings of right-sided heart failure and pulmonary hypertension. She developed status epilepticus in the setting of cardiac deterioration and respiratory infection, and died of cardiogenic and septic shock. Autopsy disclosed bilateral neuronal loss and gliosis in the inferior colliculi, locus coeruleus and facial and vestibular nuclei. Cor pulmonale is a complication of hypoventilation-induced hypoxia and hypercapnia and had not yet been reported in BVVLS.
布朗-维亚莱托-范拉埃雷综合征(BVVLS)是一种罕见的神经系统疾病,其特征是感觉神经性听力损失和多种颅神经麻痹,通常涉及第七、IX 至 XII 颅神经。我们描述了一位 33 岁女性 BVVLS 的临床和病理学特征。患者出现进行性劳累性呼吸困难,伴有右侧心力衰竭和肺动脉高压的临床和实验室发现。在心脏恶化和呼吸道感染的情况下,她发生了癫痫持续状态,并死于心源性和脓毒性休克。尸检显示下丘脑中双侧神经元丧失和神经胶质增生,蓝斑和面部及前庭核。肺心病是低通气引起的缺氧和高碳酸血症的并发症,在 BVVLS 中尚未报道。