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一个发展中国家高苯丙氨酸血症的经验:不寻常的临床表现和一种新的基因突变。

Experience with hyperphenylalaninemia in a developing country: unusual clinical manifestations and a novel gene mutation.

作者信息

Karam Pascale E, Daher Rose T, Moller Lisbeth B, Mikati Mohamad A

机构信息

Department of Pediatrics and Adolescent Medicine, American University of Beirut, Beirut, Lebanon.

出版信息

J Child Neurol. 2011 Feb;26(2):142-6. doi: 10.1177/0883073810375116. Epub 2010 Sep 7.

Abstract

We report our experience in a cohort of patients with hyperphenylalaninemia in a tertiary care referral center in Lebanon. Forty-one sequential patients were studied: 34 classical phenylketonuria (PKU), 3 hyperphenylalaninemia (non-PKU), and 4 biopterin metabolism defects. The majority of cases were clinically diagnosed at variable ages with variable neurological outcomes. Only 29.3% were detected by neonatal screening. Two unusual cases were observed in the context of inadequate treatment in 1 and delayed therapy in the other: a newborn with PKU developed severe keratomalacia; and a 5-year-old girl with dihydropteridine reductase deficiency due to a novel mutation identified in the quinoid dihydropteridine reductase gene developed Lennox-Gastaut syndrome and white matter changes with periventricular cysts. Part of our experience parallels that in the West. However, the clinical manifestations observed in our patients emphasize the importance of a national newborn screening program with efficient management of diagnosed cases.

摘要

我们报告了在黎巴嫩一家三级医疗转诊中心对一组高苯丙氨酸血症患者的治疗经验。对41例连续患者进行了研究:34例典型苯丙酮尿症(PKU)、3例高苯丙氨酸血症(非PKU)和4例生物蝶呤代谢缺陷。大多数病例在不同年龄临床诊断,神经学预后各异。仅29.3%通过新生儿筛查发现。在1例治疗不足和另1例治疗延迟的情况下观察到两例异常病例:1例PKU新生儿发生严重角膜软化症;1例5岁女孩因醌型二氢蝶呤还原酶基因中发现的新突变导致二氢蝶呤还原酶缺乏,出现Lennox-Gastaut综合征和伴有脑室周围囊肿的白质改变。我们的部分经验与西方相似。然而,我们患者中观察到的临床表现强调了开展全国新生儿筛查计划并对确诊病例进行有效管理的重要性。

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