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DOI 10.7499/j.issn.1008-8830.2019.04.014

Медицинская библиотека · Zhongguo dang dai er ke za zhi = Chinese journal of contemporary pediatrics · 2019

[Clinical features of children with lysinuric protein intolerance and SLC7A7 gene mutation: an analysis of 3 cases].

Cui Dong, Hu Yu-Hui, Tang Gen, Shen Dan, Chen Li, Liao Jian-Xiang, Chen Shu-Li

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Аннотация

Lysinuric protein intolerance (LPI) is an autosomal recessive disorder caused by SLC7A7 gene mutation and often involves severe lesions in multiple systems. Lung involvement is frequently seen in children with LPI and such children tend to have a poor prognosis. This article summarizes the clinical manifestations and gene mutation characteristics of three children diagnosed with LPI by SLC7A7 gene analysis. All three children had the manifestations of aversion to protein-rich food after weaning, delayed development, anemia, hepatosplenomegaly, and osteoporosis, as well as an increase in orotic acid in urine. In addition, interstitial pneumonia and diffuse pulmonary interstitial lesions were observed in two children. SLC7A7 gene detection showed three pathogenic mutations in these children, namely c.1387delG(p.V463CfsX56), c.1215G>A(p.W405X) and homozygous c.625+1G>A. After a definite diagnosis was made, all three children were given a low-protein diet and oral administration of citrulline [100 mg/(kg.d)], iron protein succinylate [4 mg/(kg.d)], calcium and zinc gluconates oral solution (10 mL/day) and vitamin D (400 IU/day). In addition, patient 3 was given prednisone acetate (5 mg/day). The children had varying degrees of improvement in symptoms and signs. It is hard to distinguish LPI from urea cycle disorder due to the features of amino acid and organic acid metabolism in LPI, and SLC7A7 gene analysis is the basis for a definite diagnosis of LPI.

Для цитирования: Cui Dong, Hu Yu-Hui, Tang Gen, Shen Dan, Chen Li, Liao Jian-Xiang, Chen Shu-Li. [Clinical features of children with lysinuric protein intolerance and SLC7A7 gene mutation: an analysis of 3 cases].. Zhongguo dang dai er ke za zhi = Chinese journal of contemporary pediatrics. 2019. DOI: 10.7499/j.issn.1008-8830.2019.04.014.

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