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[儿童及其患有纯合子和杂合子α1-抗胰蛋白酶缺乏症亲属的肝脏疾病]

[Liver diseases in children and their relatives with homozygous and heterozygous alpha 1-antitrypsin deficiency].

作者信息

Deutsch J, Becker H, Dubs R, Mutz I, Paschke E, Schober P, Maurer G, Fueger G F

出版信息

Wien Klin Wochenschr. 1987 Feb 6;99(3):84-91.

PMID:3495072
Abstract

Over a 6-year period 26 infants and children with homozygous (2 Z and 6 ZZ) or heterozygous alpha 1-antitrypsin deficiency (12 MZ, 6 MS) were observed prospectively and their families investigated. 7 of 8 homozygous patients had neonatal hepatitis, whereby 3 of these showed maximum transferase activities during the 5th to 9th months of life. At the age of 7 years 2 of these patients were clinically normal, but only one patient had normal transferases. One patient had cirrhosis with portal hypertension at the age of 16 years 6 months; her nephew showed hypersplenism. Family studies revealed a further 5 relatives of phenotype Z, 16 of phenotype MZ, 3 of phenotype SZ and 1 of phenotype MS; 6 of these had slightly elevated serum transferase activities. 6 patients of phenotype MZ and 2 patients of phenotype MS had neonatal hepatitis but generally with a much better prognosis than in homozygous patients. The other heterozygous patients (6 MZ and 4 MS) had a variety of additional factors determining the disease and the prognosis. Family studies showed a further 7 family members of phenotype MZ, and 2 of phenotype MS; 2 of these had slightly elevated transferase activities, 3 parents had hereditary hyperbilirubinaemia.

摘要

在6年期间,对26例患有纯合子(2例ZZ和6例Z)或杂合子α1 -抗胰蛋白酶缺乏症(12例MZ、6例MS)的婴幼儿进行了前瞻性观察,并对其家庭进行了调查。8例纯合子患者中有7例患有新生儿肝炎,其中3例在出生后第5至9个月时转氨酶活性最高。7岁时,其中2例患者临床正常,但只有1例患者的转氨酶正常。1例患者在16岁6个月时出现肝硬化并伴有门静脉高压;她的侄子出现脾功能亢进。家族研究发现了另外5例Z型表型亲属、16例MZ型表型亲属、3例SZ型表型亲属和1例MS型表型亲属;其中6例血清转氨酶活性略有升高。6例MZ型表型患者和2例MS型表型患者患有新生儿肝炎,但总体预后比纯合子患者好得多。其他杂合子患者(6例MZ和4例MS)有多种决定疾病和预后的其他因素。家族研究发现了另外7例MZ型表型家庭成员和2例MS型表型家庭成员;其中2例转氨酶活性略有升高,3例父母患有遗传性高胆红素血症

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