Shah Nikit, Lingappa Lokesh, Konanki Ramesh, Rani Sirisha, Vedam Ramprasad, Murugan Sakthivel
Department of Pediatric Neurology, Rainbow Children's Hospital, Hyderabad, Telangana, India.
Department of Pediatric Hemato-Oncology, Rainbow Children's Hospital, Hyderabad, Telangana, India.
Ann Indian Acad Neurol. 2019 Apr-Jun;22(2):231-233. doi: 10.4103/aian.AIAN_430_17.
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.
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。