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两名患有新突变的戈谢病2型患者中胎儿水肿的罕见病因。

A rare cause of hydrops fetalis in two Gaucher disease type 2 patients with a novel mutation.

作者信息

Kılavuz Sebile, Basaranoglu Murat, Epcacan Serdar, Bako Derya, Ozer Arife, Donmez Yasemin Nuran, Ceylan Emine Ipek, Tukun Ajlan, Ceylaner Serdar, Geylani Hadi, Mungan Halise Neslihan Onenli

机构信息

Division of Pediatric Metabolism and Nutrition, Department of Pediatrics, Van Training and Research Hospital, University of Health Sciences, Van, Turkey.

Department of Pharmacology, University of Oxford, Mansfield Road, Oxford, OX1 3QT, UK.

出版信息

Metab Brain Dis. 2022 Apr;37(4):1283-1287. doi: 10.1007/s11011-022-00942-5. Epub 2022 Mar 7.

Abstract

Gaucher disease type 2 is the most progressive and the rarest form of Gaucher disease, defined as the acute neuronopathic type. We presented two GD2 patients who died before three months of age due to severe septicemia, respiratory and liver failure. One was homozygous for a novel GBA variant c.590 T > A (p.197 K), and the second homozygous for the known GBA mutation c.1505G > A (p.R502H). Ichthyosis, hydrops fetalis, apnea, myoclonic seizures, and hepatosplenomegaly occurred in both patients, but hypertrophic cardiomyopathy was observed only in the second and unilateral cataract in the first patient. Due to the disease's early and rapid neurological progression, we did not administer ERT to our patients. It is strongly believed that early diagnosis is essential, and prenatal diagnosis makes genetic counselling possible for future pregnancies.

摘要

戈谢病2型是戈谢病最具进展性且最罕见的形式,被定义为急性神经病变型。我们报告了两名戈谢病2型患者,他们均在3个月龄前因严重败血症、呼吸和肝功能衰竭死亡。其中一名患者是新型GBA变异c.590 T>A(p.197 K)的纯合子,另一名是已知GBA突变c.1505G>A(p.R502H)的纯合子。两名患者均出现鱼鳞病、胎儿水肿、呼吸暂停、肌阵挛性癫痫发作和肝脾肿大,但仅第二名患者观察到肥厚型心肌病,第一名患者观察到单侧白内障。由于该疾病早期神经系统进展迅速,我们未对患者进行酶替代治疗(ERT)。人们坚信早期诊断至关重要,而产前诊断可为未来妊娠提供遗传咨询。

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