Nakane S, Shirabe S, Suenaga A, Yoshimura T, Nakamura T
First Department of Internal Medicine, Nagasaki University School of Medicine.
Rinsho Shinkeigaku. 1999 Feb-Mar;39(2-3):347-51.
We report a 49-year-old female with hereditary ceruloplasmin deficiency with hemosiderosis. There was a family history of the same symptoms; her brother showed hypoceruloplasminemia and decrease of the serum copper content. On physical examinations, dementia, dysarthria, downbeat nystagmus, sensorineural hearing disturbance, orthostatic hypotension, retinitis pigmentosa, diffuse goiter, and cerebellar ataxia were noted. Laboratory examinations disclosed leukopenia, diabetes mellitus, hypothyroidism, decrease of copper content in the serum and urine. Serum ferritin concentration was remarkably increased. Serum ceruloplasmin could not be detected. Biopsy of the liver showed that iron content in the liver was increased. On MRI study, dentate nucleus of the cerebellum, basal ganglia, and the liver showed low intensity in both T1 and T2 weighted images. A nonsense mutation in the ceruloplasmin gene was found in this patient. Systemic iron deposition and tissue damage were considered as caused by deficiency of function of ceruloplasmin as ferroxidase. To our knowledge, the characteristic combination of the clinical signs in this patient has not been reported.
我们报告了一名49岁患有遗传性铜蓝蛋白缺乏症伴血色素沉着症的女性。存在相同症状的家族病史;她的哥哥表现为低铜蓝蛋白血症和血清铜含量降低。体格检查发现痴呆、构音障碍、下跳性眼球震颤、感音神经性听力障碍、直立性低血压、色素性视网膜炎、弥漫性甲状腺肿和小脑共济失调。实验室检查显示白细胞减少、糖尿病、甲状腺功能减退、血清和尿液中铜含量降低。血清铁蛋白浓度显著升高。未检测到血清铜蓝蛋白。肝脏活检显示肝脏中铁含量增加。MRI研究显示,小脑齿状核、基底神经节和肝脏在T1和T2加权图像上均呈低信号。该患者在铜蓝蛋白基因中发现了一个无义突变。全身铁沉积和组织损伤被认为是由于铜蓝蛋白作为铁氧化酶功能缺陷所致。据我们所知,该患者临床体征的特征性组合尚未见报道。