Huh Young Eun, Kim Ji-Soo, Kim Hyo-Jung, Park Seong-Ho, Jeon Beom Seok, Kim Jong-Min, Cho Jin Whan, Zee David S
Department of Neurology, Seoul National University College of Medicine, Seoul National University Bundang Hospital, 300 Gumi-dong, Bundang-gu, Seongnam-si, Gyeonggi-do, 463-707, Korea.
Cerebellum. 2015 Jun;14(3):284-91. doi: 10.1007/s12311-015-0650-3.
In spinocerebellar ataxia type 6 (SCA6), the vestibular dysfunction and its correlation with other clinical parameters require further exploration. We determined vestibular responses over a broad range of stimulus acceleration in 11 patients with SCA6 (six men, age range=33-72 years, mean age±SD=59±12 years) using bithermal caloric irrigations, rotary chair, and head impulse tests. Correlations were also pursued among disability scores, as measured using the International Cooperative Ataxia Rating Scale, disease duration, age at onset, cytosine-adenine-guanine (CAG) repeat length, and the gain of the vestibulo-ocular reflex (VOR). In response to relatively low-acceleration, low-frequency rotational and bithermal caloric stimuli, the VOR gains were normal or increased regardless of the severity of disease. On the other hand, with relatively high-acceleration, high-frequency head impulses, there was a relative increase in gain in the mildly affected patients and a decrease in gain in the more severely affected patients and gains were negatively correlated with the severity of disease (Spearman correlation, R=-0.927, p<0.001). Selective decrease of the vestibular responses during high-acceleration, high-frequency stimuli may be ascribed to degeneration of either the flocculus or vestibular nuclei. The performance of the VOR during high-acceleration, high-frequency head impulses may be a quantitative indicator of clinical decline in SCA6.
在6型脊髓小脑共济失调(SCA6)中,前庭功能障碍及其与其他临床参数的相关性仍需进一步探索。我们使用双侧冷热试验、转椅试验和头脉冲试验,测定了11例SCA6患者(6名男性,年龄范围为33 - 72岁,平均年龄±标准差 = 59±12岁)在广泛刺激加速度范围内的前庭反应。我们还研究了使用国际合作共济失调评定量表测量的残疾评分、病程、发病年龄、胞嘧啶 - 腺嘌呤 - 鸟嘌呤(CAG)重复长度以及前庭眼反射(VOR)增益之间的相关性。对于相对低加速度、低频旋转和双侧冷热刺激,无论疾病严重程度如何,VOR增益均正常或增加。另一方面,对于相对高加速度、高频头脉冲,轻度受影响患者的增益相对增加,而重度受影响患者的增益则降低,且增益与疾病严重程度呈负相关(Spearman相关性,R = -0.927,p < 0.001)。在高加速度、高频刺激期间前庭反应的选择性降低可能归因于绒球或前庭核的退化。在高加速度、高频头脉冲期间VOR的表现可能是SCA6临床衰退的定量指标。