Fujikane M, Katayama S, Yokota N, Hirata K, Ichimaru Y
Department of Neurology, Dokkyo University, School of Medicine.
Rinsho Shinkeigaku. 1994 Apr;34(4):391-5.
The patient was a 31-year-old male who was admitted to our hospital due to slowly progressive weakness of the right upper limb and both lower limbs. Physical examination showed a tall stature, gynecomastia, feminine pubic hair, small penis and small testis. His intellectual function on the Kohs-cubic test scored 85, on the WAIS scored performance 80, verbal 74 and full scale 78. He showed BE mixed type in YG personality test and neurotic in CMI. No abnormalities were observed except for severe weakness of the right upper limb, moderate weakness of the bilateral lower limbs and hypalgesia in the limbs. Clinical laboratory tests revealed low blood and urinary testosterone levels, and a high blood gonadotropin level. Cytogenetic studies showed a 47, XXY karyotype. On neuroradiological examinations, there were small bilateral carotid canals in CT scan and multiple long T2 lesions in MRI. Angiography showed hypoplasia of the bilateral internal carotid arteries and dilatation of the bilateral vertebral arteries. The results of activity measurements of affected limbs using actigraph indicated that a conversion type hysteria had resulted in weakness of the limbs. There is no report of Klinefelter syndrome accompanied by the hypoplasia of bilateral internal carotid arteries, and the condition is considered to be extremely rare.
该患者为一名31岁男性,因右上肢和双下肢进行性缓慢无力入院。体格检查显示身材高大、乳房发育、阴毛呈女性化、阴茎小和睾丸小。他在考斯方块测验中的智力功能得分为85分,在韦氏成人智力量表中的操作智商得分为80分,言语智商得分为74分,全量表智商得分为78分。他在YG性格测试中表现为BE混合型,在CMI中表现为神经质。除右上肢严重无力、双下肢中度无力和肢体痛觉减退外,未观察到其他异常。临床实验室检查显示血睾酮和尿睾酮水平低,血促性腺激素水平高。细胞遗传学研究显示核型为47, XXY。神经放射学检查显示,CT扫描显示双侧颈动脉管小,MRI显示多个长T2信号病灶。血管造影显示双侧颈内动脉发育不全,双侧椎动脉扩张。使用活动记录仪对患侧肢体进行活动测量的结果表明,转换型癔症导致了肢体无力。目前尚无克氏综合征伴双侧颈内动脉发育不全的报道,这种情况被认为极为罕见。