Wilson C J, Aftimos S
Department of Clinical Genetics, Starship Children's Hospital, Auckland, New Zealand.
Am J Med Genet. 1998 Jul 7;78(3):300-2. doi: 10.1002/(sici)1096-8628(19980707)78:3<300::aid-ajmg19>3.0.co;2-j.
X-linked dominant chondrodysplasia punctata is characterised by resolving irregular punctate calcifications of epiphyses, variable ichthyosis and atrophoderma, short stature, and cataracts. We report on a patient with this syndrome who had transiently abnormal peroxisomal function tests. We review the literature and propose that X-linked dominant chondrodysplasia punctata is a peroxisomal disorder and that its phenotype can be explained by X chromosome lyonisation and the relative proliferation of cells expressing the normal X allele.
X连锁显性点状软骨发育不良的特征为骨骺不规则点状钙化的消退、可变的鱼鳞病和皮肤萎缩、身材矮小以及白内障。我们报告了一名患有该综合征的患者,其过氧化物酶体功能测试曾出现短暂异常。我们回顾了文献并提出,X连锁显性点状软骨发育不良是一种过氧化物酶体疾病,其表型可通过X染色体失活以及表达正常X等位基因的细胞的相对增殖来解释。