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新生儿因声带麻痹致暴发性心源性死亡,干血斑可用于死后诊断 PPA2 相关病例。

Postmortem diagnosis of PPA2-associated sudden cardiac death from dried blood spot in a neonate presenting with vocal cord paralysis.

机构信息

Rady Children's Institute of Genomic Medicine, University of California San Diego, La Jolla, California 92093, USA.

Division of Pediatric Critical Care Medicine, Department of Pediatrics, University of California San Diego, La Jolla, California 92093, USA.

出版信息

Cold Spring Harb Mol Case Stud. 2020 Oct 7;6(5). doi: 10.1101/mcs.a005611. Print 2020 Oct.

Abstract

Biallelic variants in inorganic pyrophosphatase 2 (PPA2) are known to cause infantile sudden cardiac failure (OMIM #617222), but relatively little is known about phenotypic variability of these patients prior to their death. We report a 5-wk-old male with bilateral vocal cord paralysis and hypertension who had a sudden unexpected cardiac death. Subsequently, molecular autopsy via whole-genome sequencing from newborn dried blood spot identified compound heterozygous mutations in PPA2, with a paternally inherited, pathogenic missense variant (c.514G > A; p.Glu172Lys) and a novel, maternally inherited missense variant of uncertain significance (c.442A > T; p.Thr148Ser). This report expands the presenting phenotype of patients with PPA2 variants. It also highlights the utility of dried blood spots for postmortem molecular diagnosis.

摘要

无机焦磷酸酶 2 (PPA2) 的双等位基因突变已知可导致婴儿突发性心衰竭(OMIM#617222),但在这些患者死亡之前,对其表型变异性的了解相对较少。我们报告了一例 5 周大的男性,患有双侧声带麻痹和高血压,突然发生意外心脏死亡。随后,通过对新生儿干血斑进行全基因组测序的分子尸检,在 PPA2 中发现了复合杂合突变,一个是父系遗传的致病性错义突变(c.514G > A;p.Glu172Lys),另一个是新的、母系遗传的意义不明的错义突变(c.442A > T;p.Thr148Ser)。本报告扩展了 PPA2 变异患者的表现型。它还强调了干血斑在死后分子诊断中的应用。

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