Yamana T, Murakami N, Muroga T, Sugimura K
Department of Neurology, Higashi Nagoya National Hospital.
Rinsho Shinkeigaku. 1990 Nov;30(11):1271-4.
A 19-year-old male patient with Down's syndrome accompanied by relapsing and steroid-dependent chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) was reported. He had been well until the beginning of June 1988, and he rapidly developed a gait disturbance and symmetrical numbness in his arms and legs at the end of that month. He also suffered from difficulty in swallowing and dyspnea soon afterward. A physical examination revealed the specific clinical features of Down's syndrome, but no particular signs. Neurological examination showed the following abnormal findings; fixed nystagmus, diminished gag reflex, flaccid tetraparesis, and distal dominant dysesthesia of upper and lower extremities. Deep tendon reflexes were diminished and Babinski's sign was negative. No autonomic dysfunction was detected. Routine hematology and biochemistry screening test revealed normal level except for increasing GPT and IgG. Protein and gamma globulin values in cerebrospinal fluid (CSF) were elevated without pleocytosis. Nerve conduction velocities were slighty reduced. Pulse therapy and corticosteroid treatment improved on clinical course and CSF results. CIDP probably results from an abnormal immune responsiveness. Down's syndrome has often been taken for autoimmune abnormality. It is of great interest whether there is a close connection of autoimmune abnormality between Down's syndrome and CIDP. This might be the first case report of Down's syndrome associated with CIDP, judging from our survey of the literature in Japan and other countries.
报告了一名19岁男性唐氏综合征患者,伴有复发性且依赖类固醇的慢性炎症性脱髓鞘性多发性神经根神经病(CIDP)。他在1988年6月初之前情况良好,当月月底迅速出现步态障碍以及手臂和腿部对称性麻木。此后不久他还出现吞咽困难和呼吸困难。体格检查显示出唐氏综合征的特定临床特征,但无特殊体征。神经系统检查发现以下异常:固定性眼球震颤、咽反射减弱、四肢弛缓性瘫痪以及上下肢远端为主的感觉异常。腱反射减弱,巴宾斯基征阴性。未检测到自主神经功能障碍。常规血液学和生化筛查检查除谷丙转氨酶(GPT)和免疫球蛋白G(IgG)升高外,其余指标均正常。脑脊液(CSF)中的蛋白质和γ球蛋白值升高,无细胞增多。神经传导速度略有降低。脉冲疗法和皮质类固醇治疗使临床病程和脑脊液结果有所改善。CIDP可能源于异常的免疫反应性。唐氏综合征常被认为存在自身免疫异常。唐氏综合征与CIDP之间是否存在自身免疫异常的密切联系非常值得关注。从我们对日本和其他国家文献的调查来看,这可能是首例唐氏综合征合并CIDP的病例报告。