Cerrahpasa Medical Faculty, Department of Neurology, Division of Pediatric Neurology, Istanbul University, Turkey.
Cerebellum. 2011 Mar;10(1):49-60. doi: 10.1007/s12311-010-0225-2.
Unilateral cerebellar hypoplasia (UCH) is a rare pathological condition characterized by the loss of volume in cerebellar hemispheres ranging from mild asymptomatic to severe symptomatic cases. As the designation of UCH remains problematic, the underlying etiopathogenesis also lacks explanation. We investigated the patients admitted to Departments of Child Neurology, Neurology, and Genetics between the years 1992 and 2010 and detected 12 patients with unilateral cerebellar volume loss, with the exclusion of all other cerebellar pathologies. The ages of patients ranged between 6 months to 55 years. Five patients had a delay in developmental milestones, and one of these was diagnosed with neurofibromatosis type 1. Two patients had epileptic seizures, one patient had peripheral facial paralysis as a component of Moebius syndrome, and four patients were incidentally diagnosed during etiological work-up for headache. The clinical outcomes of patients varied from healthy subjects to marked developmental impairment. Radiologically, five patients had severe disproportionate UCH, six had moderate disproportionate, and one had mild proportionate UCH. Cerebellar peduncles were affected in all, and vermis was partly hypoplastic in eight patients. Brainstem was involved in four patients, and seven patients showed involvement of white matter and/or corpus callosum. Imaging features supported that patients with severe disproportionate UCH also displayed additional cerebral and commissural changes, which were related to ischemic or vascular injuries, implying a prenatally acquired disruption. In the presence of such a wide spectrum of clinical and radiological features, a prenatally acquired lesion and, thus, a disruption seem to be more explanatory rather than a primary developmental process or malformation in the etiopathogenesis of unilateral cerebellar hypoplasia.
单侧小脑发育不全 (UCH) 是一种罕见的病理状况,其特征是小脑半球体积丧失,从轻度无症状到重度症状不等。由于 UCH 的命名仍存在问题,其潜在的病因发病机制也缺乏解释。我们调查了 1992 年至 2010 年间在儿童神经病学、神经病学和遗传学系就诊的患者,并发现了 12 例单侧小脑体积损失的患者,排除了所有其他小脑病变。患者年龄在 6 个月至 55 岁之间。5 名患者存在发育里程碑延迟,其中 1 名被诊断为神经纤维瘤病 1 型。2 名患者出现癫痫发作,1 名患者患有作为 Moebius 综合征组成部分的周围性面瘫,4 名患者在头痛病因学检查中意外诊断。患者的临床结局从健康到明显的发育障碍不等。影像学上,5 名患者存在严重的不成比例 UCH,6 名患者存在中度不成比例 UCH,1 名患者存在轻度比例 UCH。所有患者的小脑脚均受影响,8 名患者的蚓部部分发育不全。4 名患者累及脑干,7 名患者的白质和/或胼胝体受累。影像学特征表明,严重不成比例 UCH 的患者还存在额外的大脑和连合变化,这与缺血或血管损伤有关,表明其在产前发生。在存在如此广泛的临床和影像学特征的情况下,产前获得的损伤,因此,中断似乎比单侧小脑发育不全的病因发病机制中的原发性发育过程或畸形更具解释性。