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一个具有显性遗传性耳聋-甲营养不良(DDOD)综合征的家族的报告。

Report of a further family with dominant deafness-onychodystrophy (DDOD) syndrome.

机构信息

Genetic Health Services Victoria, Royal Children’s Hospital, Parkville, Australia.

出版信息

Am J Med Genet A. 2011 Oct;155A(10):2512-5. doi: 10.1002/ajmg.a.34184.

Abstract

Deafness and onychodystrophy is a presumed autosomal dominant condition previously reported in five families. We report on a three-generation family with three affected members with absent finger and toenails, finger-like thumbs, and severe sensorineural deafness. In the hands, the first and fifth digits had absent nails and bulbous swelling of the distal phalanx. The second, third, and fourth digits were relatively spared. In the feet,there were hypoplastic great toenails and absent nails on second to fifth toes. Intelligence was normal. In addition to deafness and onychodystrophy, other features in this family included subtle facial dysmorphism in two individuals, cutis aplasia in one individual, epilepsy in one individual, and sudden infant death in two family members, one of whom had deafness and onychodystrophy and one who did not. A full autopsy of both infants did not reveal a cause. SNP microarray analysis of one family member showed no evidence of copy number change. This family's condition fits within the spectrum of dominant deafness-onychodystrophy syndrome (DDOD) and further characterises this rare condition.

摘要

耳聋和甲营养不良是一种假定的常染色体显性遗传疾病,先前已在五个家族中报道过。我们报告了一个三代同堂的家族,有三个受影响的成员,他们存在无指(趾)甲、拇指状似手指、严重的感觉神经性耳聋。在手部,第一和第五指没有指甲,末节指骨呈球状肿胀。第二、第三和第四指相对幸免。在足部,大脚趾甲发育不良,第二至第五脚趾无指甲。智力正常。除了耳聋和甲营养不良,这个家族的其他特征还包括两个人有轻微的面部畸形,一个人有表皮发育不全,一个人有癫痫,两个家庭成员猝死,其中一个有耳聋和甲营养不良,另一个没有。对两个婴儿进行的全面尸检没有发现死因。对一个家族成员的 SNP 微阵列分析显示没有拷贝数变化的证据。这个家族的情况符合显性耳聋-甲营养不良综合征(DDOD)的范畴,并进一步描述了这种罕见的情况。

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