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碳水化合物代谢中的遗传紊乱:与乳糜泻相关的遗传性果糖不耐受症。

Genetic disorder in carbohydrates metabolism: hereditary fructose intolerance associated with celiac disease.

作者信息

Păcurar Daniela, Leşanu Gabriela, Dijmărescu Irina, Ţincu Iulia Florentina, Gherghiceanu Mihaela, Orăşeanu Dumitru

机构信息

Emergency Department, "Grigore Alexandrescu" Emergency Hospital for Children, Bucharest, Romania;

出版信息

Rom J Morphol Embryol. 2017;58(3):1109-1113.

Abstract

Celiac disease (CD) has been associated with several genetic and immune disorders, but association between CD and hereditary fructose intolerance (HFI) is extremely rare. HFI is an autosomal recessive disease caused by catalytic deficiency of aldolase B (fructose-1,6-bisphosphate aldolase). We report the case of a 5-year-old boy suffering from CD, admitted with an initial diagnosis of Reye's-like syndrome. He presented with episodic unconsciousness, seizures, hypoglycemia, hepatomegaly and abnormal liver function. The patient has been on an exclusion diet for three years, but he still had symptoms: stunting, hepatomegaly, high transaminases, but tissue transglutaminase antibodies were negative. Liver biopsy showed hepatic steatosis and mitochondrial damage. The dietary history showed an aversion to fruits, vegetables and sweet-tasting foods. The fructose tolerance test was positive, revealing the diagnostic of hereditary fructose intolerance. Appropriate dietary management and precautions were recommended. The patient has been symptom-free and exhibited normal growth and development until 10 years of age.

摘要

乳糜泻(CD)与多种遗传和免疫疾病相关,但CD与遗传性果糖不耐受(HFI)之间的关联极为罕见。HFI是一种常染色体隐性疾病,由醛缩酶B(果糖-1,6-二磷酸醛缩酶)催化缺陷引起。我们报告了一名5岁患乳糜泻男孩的病例,最初诊断为类瑞氏综合征入院。他表现为发作性意识丧失、癫痫发作、低血糖、肝肿大和肝功能异常。该患者已进行三年的排除饮食,但仍有症状:发育迟缓、肝肿大、转氨酶升高,但组织转谷氨酰胺酶抗体呈阴性。肝脏活检显示肝脂肪变性和线粒体损伤。饮食史显示其厌恶水果、蔬菜和甜味食物。果糖耐量试验呈阳性,确诊为遗传性果糖不耐受。建议采取适当的饮食管理和预防措施。该患者直到10岁一直无症状,生长发育正常。

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