Lungarotti M S, Martello C, Barboni G, Mezzetti D, Mariotti G, Calabro A
Istituto di Pediatria, Universitá di Perugia, Italy.
Am J Med Genet. 1994 Jul 15;51(4):598-601. doi: 10.1002/ajmg.1320510460.
We report on 2 patients with mental retardation and bullous dystrophy, macular type. The observation of the condition in a male and his maternal uncle is consistent with recessive X-linkage. Due to the rarity of the condition, nosologic definition was difficult before the birth of the propositus. The clinical picture in the two patients described, characterized by mental retardation, dwarfism, microcephaly, alopecia, bullous dystrophy macular type, hypogenitalism, is very much like the one observed in the patients, all males, belonging to the only other family reported to date. The recent localization of the bullous dystrophy gene in the Xq24-qter segment opens the possibility of prenatal diagnosis.
我们报告了2例患有智力发育迟缓及大疱性营养不良(黄斑型)的患者。在一名男性患者及其舅舅身上观察到这种病症,这与隐性X连锁遗传相符。由于该病症罕见,在先证者出生前很难进行疾病分类定义。所描述的这两名患者的临床表现为智力发育迟缓、侏儒症、小头畸形、脱发、黄斑型大疱性营养不良、生殖器发育不全,与迄今为止报道的唯一另一个家族中的所有男性患者所观察到的症状非常相似。最近大疱性营养不良基因定位在Xq24-qter区段,这为产前诊断提供了可能。