Vanikieti Kavin, Cheecharoen Piyaphon, Jindahra Panitha, Lueangaram Sirin, Padungkiatsagul Tanyatuth
Department of Ophthalmology.
Department of Radiology.
Int Med Case Rep J. 2016 Sep 6;9:273-7. doi: 10.2147/IMCRJ.S109357. eCollection 2016.
Vertebrobasilar dolichoectasia (VBD) is a rare dilative arteriopathy defined as elongation or widening of the intracranial vertebral and/or basilar arteries. The prevalence ranges from 0.06% to 5.8%. The majority of VBDs are asymptomatic. Downbeat nystagmus has been reported as a component of an infrequent ocular movement disorder in VBD. Nevertheless, oculopalatal tremor (OPT), delayed sequelae of a brainstem lesion, has never been demonstrated in VBD cases. Synchronized rhythmic involuntary contractions of the soft palate with an ocular pendular nystagmus, predominantly vertical pendular nystagmus, are the hallmark presentation. Our case demonstrated a 50-year-old female who presented with 3-month history of oscillopsia. Examination showed binocular conjugate torsional jerk nystagmus simultaneous with a contraction of the soft palate, defined as an atypical OPT, resulting from dolichoectatic left vertebral artery compressing on medulla, demonstrated by magnetic resonance imaging. Bilateral conjugate torsional jerk nystagmus simultaneous with palatal tremor, considering as an atypical OPT, should be included as one of the symptomatic presentations of vertebral artery dolichoectasia in spite of its rarity.
椎基底动脉延长扩张症(VBD)是一种罕见的扩张性动脉病变,定义为颅内椎动脉和/或基底动脉的延长或增宽。患病率在0.06%至5.8%之间。大多数VBD患者无症状。下跳性眼球震颤已被报道为VBD中一种罕见的眼球运动障碍的组成部分。然而,眼腭震颤(OPT)作为脑干病变的延迟后遗症,在VBD病例中从未得到证实。软腭同步节律性不自主收缩伴眼球摆动性眼球震颤,主要是垂直摆动性眼球震颤,是其标志性表现。我们的病例是一名50岁女性,有3个月的视振荡病史。检查显示双眼共轭扭转性急跳性眼球震颤,同时软腭收缩,定义为非典型OPT,磁共振成像显示是由延长扩张的左椎动脉压迫延髓所致。尽管罕见,但双侧共轭扭转性急跳性眼球震颤伴腭部震颤,考虑为非典型OPT,应被列为椎动脉延长扩张症的症状性表现之一。