Benson P F, Button L R, Fensom A H, Dean M F
Clin Genet. 1979 Nov;16(5):317-22. doi: 10.1111/j.1399-0004.1979.tb01009.x.
Although radiological involvement of the lower dorsal and upper lumbar vertebrae is common in the severe form of Hunter's disease (MPS II), there are reports in the literature that clinical kyphosis does not occur. We report a boy with marked clinical kyphosis in whom the diagnosis of MPS II was proved by demonstrating a severe deficiency of serum and leucocyte iduronate-sulphate sulphatase and an accelerated incorporation of radiosulphate into his cultured fibroblast glycosaminoglycans, which could not be corrected by the product of other typed reference MPS II cells. The existence of several other genetic diseases, sometimes complicated by kyphosis, was excluded by assay of fibroblast lysosomal enzymes.
尽管在严重型亨特氏病(黏多糖贮积症II型)中,下胸椎和上腰椎的放射学受累很常见,但文献中有报道称并未出现临床脊柱后凸。我们报告了一名有明显临床脊柱后凸的男孩,通过检测发现其血清和白细胞艾杜糖醛酸硫酸酯酶严重缺乏,且放射性硫酸盐加速掺入其培养的成纤维细胞糖胺聚糖中,而其他类型的黏多糖贮积症II型参考细胞的产物无法纠正这一情况,从而证实了黏多糖贮积症II型的诊断。通过检测成纤维细胞溶酶体酶,排除了其他几种有时并发脊柱后凸的遗传疾病。