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一名印度婴儿因嘌呤核苷磷酸化酶基因新突变导致免疫缺陷、运动发育迟缓及低尿酸血症

Immunodeficiency, Motor Delay, and Hypouricemia Caused by a Novel Mutation of Purine Nucleoside Phosphorylase Gene in an Indian Infant.

作者信息

Shah Nikit, Lingappa Lokesh, Konanki Ramesh, Rani Sirisha, Vedam Ramprasad, Murugan Sakthivel

机构信息

Department of Pediatric Neurology, Rainbow Children's Hospital, Hyderabad, Telangana, India.

Department of Pediatric Hemato-Oncology, Rainbow Children's Hospital, Hyderabad, Telangana, India.

出版信息

Ann Indian Acad Neurol. 2019 Apr-Jun;22(2):231-233. doi: 10.4103/aian.AIAN_430_17.

Abstract

We describe an 11-month-old boy who presented with recurrent respiratory infections from 6 months of age. His elder sister died at 10 months with severe septicemia and meningitis. The boy had a mild motor delay. Investigations revealed T cell deficiency and very low serum uric acid suggestive of purine nucleoside phosphorylase (PNP) deficiency - a rare variant of severe combined immunodeficiency disease. A novel homozygous missense mutation of c.597C>G(p. S199R) of exon 5 on PNP gene confirmed the diagnosis. We suggest that uric acid should be a part of investigation profile for unidentified motor delay, as recurrent infections can be late presentation.

摘要

我们描述了一名11个月大的男孩,他从6个月大起就反复出现呼吸道感染。他的姐姐在10个月大时死于严重败血症和脑膜炎。该男孩有轻度运动发育迟缓。检查发现T细胞缺陷以及血清尿酸极低,提示嘌呤核苷磷酸化酶(PNP)缺乏——这是重症联合免疫缺陷病的一种罕见变体。PNP基因第5外显子的c.597C>G(p.S199R)新型纯合错义突变确诊了该诊断。我们建议,对于不明原因的运动发育迟缓,尿酸应作为检查项目的一部分,因为反复感染可能是较晚出现的症状。

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Purine nucleoside phosphorylase deficiency: a mutation update.
Nucleosides Nucleotides Nucleic Acids. 2011 Dec;30(12):1243-7. doi: 10.1080/15257770.2011.630852.
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Persistent developmental delay despite successful bone marrow transplantation for purine nucleoside phosphorylase deficiency.
J Pediatr Hematol Oncol. 2002 Jan;24(1):69-71. doi: 10.1097/00043426-200201000-00018.

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