病例报告:戈谢病的诊断在幼儿急性呼吸衰竭。

IF 2.1 3区 医学 Q2 PEDIATRICS
Frontiers in Pediatrics Pub Date : 2025-03-14 eCollection Date: 2025-01-01 DOI:10.3389/fped.2025.1476541
Sarah Householder, Ruchit Nagar, Nisarg Shah, Jodi Forward, Sean Bickerton, Pramod Mistry, E Vincent S Faustino
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引用次数: 0

摘要

一个22个月大的男婴在轻微跌倒后表现为发绀和喘鸣,然后发展为急性呼吸窘迫。患者的呼吸状况迅速发展为严重急性呼吸窘迫综合征。脾功能亢进的其他发现促使综合多学科方法和考虑先天性代谢错误。快速全基因组测序显示GBA1基因存在复合杂合子突变,涉及母体遗传的已知致病变异p.L484P和父亲遗传的新型可能致病变异p.P358l。白细胞酸β-葡萄糖苷酶活性低,确诊为戈谢病,患者接受重组巨噬细胞靶向酶替代治疗。患者最终康复,但随后的检查显示严重的球功能障碍,并有误吸的证据。出院两个月后,患者因疑似误吸并咯血而出现心脏骤停。本病例说明了先前未描述的戈谢病的表现和一种新的可能的戈谢病致病基因变异。它强调了多学科方法的作用,包括快速全基因组测序,以建立及时诊断和提供适当的治疗戈谢病。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Case Report: Diagnosis of Gaucher disease in a toddler with acute respiratory failure.

A 22-month-old male infant presented with cyanosis and stridor after a trivial fall and then developed acute respiratory distress. The respiratory status of the patient progressed rapidly to severe acute respiratory distress syndrome. Additional findings of hypersplenism prompted a comprehensive multidisciplinary approach and consideration of an inborn error of metabolism. Rapid whole-genome sequence showed a compound heterozygote mutation in the GBA1 gene involving a maternally inherited known pathogenic variant, p.L484P, and a paternally inherited novel likely pathogenic variant, p.P358l. The diagnosis of Gaucher disease was confirmed with low leukocyte acid β-glucosidase activity and the patient received recombinant macrophage-targeted enzyme replacement therapy. The patient eventually recovered, but subsequent work-up demonstrated severe bulbar dysfunction with evidence of aspiration. Two months after discharge, the patient arrived at the hospital in a condition of cardiac arrest after a suspected aspiration event associated with hemoptysis. This case illustrates a previously undescribed presentation of Gaucher disease and a new likely pathogenic genetic variant for Gaucher disease. It highlights the role of a multidisciplinary approach, including rapid whole-genome sequencing, to establish timely diagnosis and provide appropriate therapy for Gaucher disease.

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来源期刊
Frontiers in Pediatrics
Frontiers in Pediatrics Medicine-Pediatrics, Perinatology and Child Health
CiteScore
3.60
自引率
7.70%
发文量
2132
审稿时长
14 weeks
期刊介绍: Frontiers in Pediatrics (Impact Factor 2.33) publishes rigorously peer-reviewed research broadly across the field, from basic to clinical research that meets ongoing challenges in pediatric patient care and child health. Field Chief Editors Arjan Te Pas at Leiden University and Michael L. Moritz at the Children''s Hospital of Pittsburgh are supported by an outstanding Editorial Board of international experts. This multidisciplinary open-access journal is at the forefront of disseminating and communicating scientific knowledge and impactful discoveries to researchers, academics, clinicians and the public worldwide. Frontiers in Pediatrics also features Research Topics, Frontiers special theme-focused issues managed by Guest Associate Editors, addressing important areas in pediatrics. In this fashion, Frontiers serves as an outlet to publish the broadest aspects of pediatrics in both basic and clinical research, including high-quality reviews, case reports, editorials and commentaries related to all aspects of pediatrics.
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