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遗传性血色素沉着症:从基因到临床

Inherited hemochromatosis: from genetics to clinics.

作者信息

Camaschella C, Merlini R

机构信息

Department of Clinical and Biological Sciences, University of Turin, San Luigi Hospital, Orbassano (Turin), Italy.

出版信息

Minerva Med. 2005 Jun;96(3):207-22.

Abstract

Hereditary hemochromatosis is one of the most common autosomal recessive disorder among Caucasians since the genotype at risk for hemochromatosis accounts for 1:200-400 individuals of Northern European ancestry. The disease is characterized by an inappropriately increased intestinal iron absorption leading to early abnormalities of iron parameters followed by iron deposition in different organs. Excessive iron causes tissue damage and fibrosis, leading to organ failure. Clinical complications appear late in life and include liver cirrhosis, diabetes, cardiomyopathy, hypogonadism, arthropathy, skin pigmentation and susceptibility to liver cancer. Clinical symptoms develop only in homozygotes. Heterozy-gotes may show abnormalities of iron parameters, but are not clinically affected, unless carriers of other conditions which modify iron metabolism, such as chronic liver diseases, beta-thalassemia trait or other haemolytic anemias. The phenotypic expression of the disease is variable even within the same family, due to the effect of modifier genes or to environmental factors. Recent progress of genetics and molecular biology have shown that hemochromatosis is an heterogeneous disease, that may result from the inactivation of different genes. The identification of mutations of HFE and of other genes involved in the disease has allowed to develop molecular tests to support early diagnosis, allowing also to ameliorate the differential diagnosis with other iron loading disorders. In addition, the increased knowledge acquired from the study of hemochromatosis has contributed to clarify the pathophysiology of iron metabolism. For this reason hemochromatosis is considered a typical example of molecular medicine.

摘要

遗传性血色素沉着症是白种人中最常见的常染色体隐性疾病之一,因为血色素沉着症的风险基因型在北欧血统的人群中占比为1:200 - 400。该疾病的特征是肠道铁吸收异常增加,导致铁参数早期异常,随后铁在不同器官中沉积。过量的铁会导致组织损伤和纤维化,进而导致器官衰竭。临床并发症在生命后期出现,包括肝硬化、糖尿病、心肌病、性腺功能减退、关节病、皮肤色素沉着以及易患肝癌。临床症状仅在纯合子中出现。杂合子可能会出现铁参数异常,但在临床上没有影响,除非是其他改变铁代谢的疾病的携带者,如慢性肝病、β地中海贫血特征或其他溶血性贫血。由于修饰基因的作用或环境因素,即使在同一家族中,该疾病的表型表达也存在差异。遗传学和分子生物学的最新进展表明,血色素沉着症是一种异质性疾病,可能由不同基因的失活引起。HFE基因及其他与该疾病相关基因的突变的鉴定,使得能够开展分子检测以支持早期诊断,同时也有助于改善与其他铁过载疾病的鉴别诊断。此外,从血色素沉着症研究中获得的更多知识有助于阐明铁代谢的病理生理学。因此,血色素沉着症被认为是分子医学的一个典型例子。

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