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无β脂蛋白血症患者成功自然受孕:阿姆斯特丹经验还是维生素替代疗法的艺术?

A successful spontaneous pregnancy in abetalipoproteinemia: Amsterdam or the art of vitamin replacement?

作者信息

Ferreira Francisca, Patel Vinod, Matts Suzy

机构信息

Department of Acute Medicine, George Eliot Hospital, Nuneaton, UK.

Warwick Medical School University of Warwick, Coventry, UK Departments of Endocrinology and Diabetes, General Internal Medicine, Medical Obstetrics, George Eliot Hospital NHS Trust, Nuneaton, UK.

出版信息

BMJ Case Rep. 2014 Dec 8;2014:bcr2014206754. doi: 10.1136/bcr-2014-206754.

Abstract

Abetalipoproteinemia is a rare metabolic disorder that causes disturbed lipid absorption with consequent hypocholesterolaemia and liposoluble avitaminosis. The broad spectrum of presentations includes malabsorption, failure to thrive and acanthocytosis in children, while later in life expected manifestations include coagulopathy, myopathy, retinitis pigmentosa, peripheral neuropathy, hyporeflexia and ataxia. These neurological complications stem from demyelination secondary to vitamin E deficiency. Another complication is reduced fertility in women. In the event of a successful conception, issues arise in vitamin supplementation, the mainstay of treatment of abetalipoproteinemia. The skilful clinician must master the delicate balance between the teratogenic effects on the fetus of over as well as under replacement of vitamins, pregnancy complications such as premature rupture of membranes and eclampsia, and, finally, maternal complications such as corneal ulcers. We describe the management of a patient ranging from pubertal growth to bearing a successful spontaneous pregnancy with an outcome of a completely healthy mother and child.

摘要

无β脂蛋白血症是一种罕见的代谢紊乱疾病,会导致脂质吸收障碍,进而引发低胆固醇血症和脂溶性维生素缺乏症。其临床表现多样,儿童期包括吸收不良、发育不良和棘红细胞增多症,而在成年期,预期表现包括凝血病、肌病、色素性视网膜炎、周围神经病变、反射减退和共济失调。这些神经并发症源于维生素E缺乏继发的脱髓鞘。另一个并发症是女性生育能力下降。如果成功受孕,在维生素补充方面会出现问题,而维生素补充是无β脂蛋白血症治疗的主要手段。经验丰富的临床医生必须掌握好维生素补充过量和不足对胎儿的致畸作用、胎膜早破和子痫等妊娠并发症以及角膜溃疡等母体并发症之间的微妙平衡。我们描述了一名患者从青春期发育到成功自然受孕并产下完全健康的母婴的治疗过程。

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