Hashimoto K, Takeuchi A, Ieshima A, Takada M, Kasagi M
Division of Child Neurology, Tottori University School of Medicine, Japan.
Am J Med Genet. 1994 Feb 1;49(3):266-9. doi: 10.1002/ajmg.1320490304.
We report on a 16-year-old girl with spondyloepiphyseal dysplasia, nephrotic syndrome, lymphopenia, and signs of defective cellular immunity. The manifestations are very similar to those reported by Spranger et al. [1991: J. Pediatr 119: 64-72] as Schimke immunoosseous dysplasia, except for age of onset. In Schimke immunoosseous dysplasia, growth retardations as an initial symptom is noted in early childhood and about 1 year after onset of progressive proteinuria. In our case the skeletal abnormality was noted at age 10 years as dislocation of the hip joints and the diagnosis of nephrotic syndrome was made at age 16 years. The findings strongly suggest that our patient has a juvenile variant of Schimke immunoosseous dysplasia.
我们报告了一名16岁女孩,她患有脊椎骨骺发育不良、肾病综合征、淋巴细胞减少症以及细胞免疫缺陷的体征。其表现与Spranger等人[1991年:《儿科学杂志》119卷:64 - 72页]报道的Schimke免疫骨发育不良非常相似,只是发病年龄不同。在Schimke免疫骨发育不良中,生长发育迟缓作为初始症状在幼儿期出现,且在进行性蛋白尿发作约1年后出现。在我们的病例中,10岁时发现骨骼异常,表现为髋关节脱位,16岁时诊断为肾病综合征。这些发现强烈提示我们的患者患有Schimke免疫骨发育不良的青少年型变体。