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宫内半乳糖血症的影响。

Effects of galactosemia in utero.

作者信息

Holton J B

机构信息

Department of Child Health, Royal Hospital for Sick Children, Bristol, UK.

出版信息

Eur J Pediatr. 1995;154(7 Suppl 2):S77-81. doi: 10.1007/BF02143809.

DOI:10.1007/BF02143809
PMID:7671971
Abstract

There is direct evidence that in galactosemia, due to galactose-1-phosphate uridyltransferase deficiency, galactose, galactose-1-phosphate and galactitol accumulate in the fetus by week 20 of gestation. The metabolic abnormality may develop earlier than this, however, since the key enzymes in galactose metabolism have been shown to be present in normal fetal liver from the 10th week of gestation. There has been a single report of increased galactitol in amniotic fluid obtained at 10 weeks gestation, the outcome being a baby affected with galactosemia. Cataract formation in the fetus is rare and the only direct evidence that galactosemia may have harmful effects in utero. However, it has been concluded that the liver pathology seen in some patients who died in the neonatal period originated prenatally, and some studies have found that galactosemia is associated with reduced birth weight. Reports of two patients with histologically normal ovaries very soon after birth have been cited as evidence against gonadal dysfunction arising during fetal life, but it should be noted that this is not a constant feature in female galactosemics. Other observations, particularly those made from animal models, would suggest that ovarian dysfunction is most likely to have been caused in utero.

摘要

有直接证据表明,在半乳糖血症中,由于1-磷酸半乳糖尿苷转移酶缺乏,在妊娠20周时胎儿体内会积聚半乳糖、1-磷酸半乳糖和半乳糖醇。然而,代谢异常可能比这更早出现,因为已证明从妊娠第10周起,正常胎儿肝脏中就存在半乳糖代谢的关键酶。有一份报告称,在妊娠10周时获取的羊水中半乳糖醇增加,结果娩出一名患半乳糖血症的婴儿。胎儿白内障形成很少见,是半乳糖血症可能在子宫内产生有害影响的唯一直接证据。然而,已经得出结论,一些在新生儿期死亡的患者所出现的肝脏病理变化始于产前,而且一些研究发现半乳糖血症与出生体重降低有关。有两名出生后不久卵巢组织学正常的患者报告被引为反对胎儿期出现性腺功能障碍的证据,但应指出,这并非女性半乳糖血症患者的恒定特征。其他观察结果,特别是来自动物模型的观察结果,表明卵巢功能障碍很可能是在子宫内造成的。

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本文引用的文献

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Galactose-1-phosphate in the pathophysiology of galactosemia.半乳糖-1-磷酸在半乳糖血症病理生理学中的作用
Eur J Pediatr. 1995;154(7 Suppl 2):S45-9. doi: 10.1007/BF02143803.
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