Symptomatic unruptured capillary telangiectasia of the brain stem: report of three cases and review of the literature

Symptomatic unruptured capillary telangiectasia of the brain stem: report of three cases and review of the literature
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症状性脑干未破裂毛细血管扩张症三例报道并文献复习

DOI:
10.1136/jnnp.71.3.390
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发表时间:
2001
期刊:
Journal of Neurology, Neurosurgery & Psychiatry
影响因子:
--
通讯作者:
Mario Mascalchi
Mario Mascalchi
中科院分区:
--
文献类型:
--
作者:
Cesa Scaglione;Fabrizio Salvi;P. Riguzzi;M. Vergelli;Carlo Alberto Tassinari;Mario Mascalchi

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本文报告3例有短暂或间歇性局灶性神经体征提示脑干受累的年轻患者,其中高场MRI显示T2加权自旋回波显灶区高信号,T2*加权梯度回波显低信号,增强后T1加权显强化,与未破裂的脑干毛细血管扩张相一致。第一例患者为28岁女性,主诉左耳反复耳鸣,在月经期间加重;核磁共振显示血管异常累及左侧听觉通路。第二例为30岁女性,分娩4周后出现3次阵发性左唇运动;核磁共振显示右侧皮质核通路有毛细血管扩张。第三例患者,一名36岁的男性,有一过性右侧贝尔麻痹;核磁共振显示左侧皮质脊髓束和内侧纵束有两个与毛细血管扩张一致的界限区域。毛细血管扩张性血管壁中的 类固醇受体可能是我们的两名女性患者复发和短暂病程的原因。 对毛细血管扩张症的磁共振特征的认识可能有助于确定这些血管畸形的真实发生率、临床相关性和出血并发症的风险。
Three young patients with transient or intermittent focal neurological signs suggesting brain stem involvement are described, in whom high field MRI showed focal areas of hyperintensity in T2 weighted spin echo images, hypointensity in T2* weighted gradient echo images, and enhancement in postcontrast T1 weighted images consistent with unruptured capillary telangiectasia of the brain stem. The first patient was a 28 year old woman who complained of recurrent left ear tinnitus, exacerbated during the menstrual period; MRI demonstrated that the vascular anomaly involved the left acoustic pathway. The second patient was a 30 year old woman who had three episodes of paroxysmal left lip movement 4 weeks after child delivery; MRI showed capillary telangiectasia in the right corticonuclear pathway. The third patient, a 36 year old man, had a transient right Bell's palsy; MRI disclosed two circumscribed areas consistent with capillary telangiectasia in the left corticospinal tract and medial longitudinal fasciculus.  Steroid receptors in the telangiectatic vessels walls might account for the recurrent and transient course seen in our two female patients.  Awareness of the MRI features of capillary telangiectasia may help in defining the real incidence, clinical correlation, and the risk of haemorrhagic complications of these vascular malformations.