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一种具有类矮妖精貌特征的全身性弹性纤维缺乏综合征:一种具有常染色体隐性遗传模式的独特遗传病。

A syndrome of generalized elastic fiber deficiency with leprechaunoid features: a distinct genetic disease with an autosomal recessive mode of inheritance.

作者信息

Dallaire L, Cantin M, Melançon S B, Perreault G, Potier M

出版信息

Clin Genet. 1976 Jul;10(1):1-11. doi: 10.1111/j.1399-0004.1976.tb00001.x.

Abstract

Three male infants with generalized elastolysis and leprechaunoid features from two related and consanguineous parents of Italian origin died in the first year of life following severe cardio-pulmonary complications. While these children showed a decrease in elastic fibers, no degeneration was noted and histochemical as well as systemic metabolic studies were negative. It is postulated that this disease is a variant of cutis laxa or at least that the absence of granular degeneration of the elastic fibers described by Goltz is a secondary manifestation present only in older children. Prenatal diagnosis of this syndrome is not yet possible since no intracellular or biochemical changes have been identified. In view of the familial occurrence of this syndrome, and the association of specific clinical and pathological findings, we suggest that we are dealing with a distinct hereditary disorder of the connective tissue.

摘要

三名男婴患有全身性弹性组织溶解症,并具有类矮小妖精貌特征,其父母为来自意大利的两个有血缘关系的近亲。这三名男婴在出生后第一年因严重心肺并发症死亡。虽然这些患儿的弹性纤维数量减少,但未发现变性,组织化学及全身代谢研究结果均为阴性。据推测,这种疾病是皮肤松弛症的一种变体,或者至少戈尔茨所描述的弹性纤维无颗粒变性是仅在大龄儿童中出现的继发性表现。由于尚未发现细胞内或生化变化,目前尚无法对该综合征进行产前诊断。鉴于该综合征具有家族性发病特点,以及特定临床和病理表现之间的关联,我们认为这是一种独特的遗传性结缔组织疾病。

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