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与成人罕见及严重感染相关的甘露糖结合蛋白基因突变。

Mannose binding protein gene mutations associated with unusual and severe infections in adults.

作者信息

Summerfield J A, Ryder S, Sumiya M, Thursz M, Gorchein A, Monteil M A, Turner M W

机构信息

Department of Medicine, St Mary's Hospital Medical School, Imperial College of Science, Technology and Medicine, London.

出版信息

Lancet. 1995 Apr 8;345(8954):886-9. doi: 10.1016/s0140-6736(95)90009-8.

Abstract

A defect in opsonisation can cause a common immunodeficiency. A mutation in mannose binding protein (MBP) caused by point mutations in the MBP gene will lead to such a defect. This type of syndrome can cause recurrent infections in infants between 6 and 18 months of age but is not generally believed to predispose to adult infections. We looked at 4 patients with severe and unusual infections in whom MBP gene mutations were the only identified cause of immunodeficiency and one patient with combined MBP and IgA deficiency. We analysed the MBP genotypes of all the patients in whom we suspected an immunodeficiency because of their clinical history. Infections seen were recurrent skin abscesses, chronic cryptosporidial diarrhoea, meningococcal meningitis with recurrent herpes simplex, and fatal klebsiella lobar pneumonia. Both sexes were affected and ages ranged from 15 to 56 years. Two patients were homozygous for codon 54 mutations, one patient had codon 52 and codon 54 mutations and was phenotypically homozygous, and two patients were heterozygous for codon 54 mutations. Individuals homozygous for MBP mutations are unusual in the general population (approximate frequency 0.3%). The occurrence of three homozygotes for MBP mutations among these five infected patients suggests that MBP deficiency may confer a life-long risk of infection.

摘要

调理作用缺陷可导致一种常见的免疫缺陷。由甘露糖结合蛋白(MBP)基因中的点突变引起的MBP突变会导致此类缺陷。这种综合征可导致6至18个月大的婴儿反复感染,但一般认为不会引发成人感染。我们研究了4例患有严重且不寻常感染的患者,其中MBP基因突变是唯一确定的免疫缺陷原因,还有1例患有MBP和IgA联合缺陷的患者。我们分析了所有因临床病史而怀疑存在免疫缺陷的患者的MBP基因型。观察到的感染包括反复出现的皮肤脓肿、慢性隐孢子虫腹泻、伴有复发性单纯疱疹的脑膜炎球菌性脑膜炎以及致命的克雷伯菌大叶性肺炎。男女均受影响,年龄范围为15至56岁。两名患者为密码子54突变的纯合子,一名患者有密码子52和密码子54突变且表型为纯合子,两名患者为密码子54突变的杂合子。MBP突变纯合子个体在一般人群中较为罕见(大致频率为0.3%)。这5名感染患者中有3名是MBP突变的纯合子,这表明MBP缺陷可能会带来终身感染风险。

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