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以持续性新生儿呼吸窘迫为表现的I型黏多糖贮积症:一例报告

Mucopolysaccharidosis Type I Presenting with Persistent Neonatal Respiratory Distress: A Case Report.

作者信息

Asseri Ali Alsuheel, Alzoani Ahmad, Almazkary Abdulwahab M, Abdulaziz Nisreen, Almazkary Mufareh H, Alahmari Samy Ailan, Duraisamy Arul J, Sureshkumar Shruti

机构信息

Department of Child Health, King Khalid University, Abha 62529, Saudi Arabia.

Department of Neonatology, Abha Maternity and Children Hospital, Ministry of Health, Abha 62521, Saudi Arabia.

出版信息

Diseases. 2023 Apr 28;11(2):67. doi: 10.3390/diseases11020067.

Abstract

Mucopolysaccharidosis type I (MPS I) is a rare inherited autosomal recessive lysosomal storage disorder. Despite several reports on MPS I-related neonatal interstitial lung disease, it is still considered to be an under-recognized disease manifestation. Thus, further study of MPS I is required to improve specific therapies and management strategies. The current report describes a late preterm baby (36 weeks gestational age) with neonatal onset of interstitial lung disease eventually diagnosed as MPS I. The neonate required prolonged respiratory support and oxygen supplementation that further escalated the likely diagnosis of inherited disorders of pulmonary surfactant dysfunction. Whole-exome sequencing confirmed the diagnosis of MPS I, following the observation of low levels of the enzyme α-L-iduronidase. The results highlight the necessity of considering MPS I-related pulmonary involvement in newborns with persistent respiratory insufficiency.

摘要

I型黏多糖贮积症(MPS I)是一种罕见的常染色体隐性遗传性溶酶体贮积病。尽管已有多篇关于MPS I相关新生儿间质性肺病的报道,但它仍被认为是一种未得到充分认识的疾病表现。因此,需要进一步研究MPS I以改进特异性治疗方法和管理策略。本报告描述了一名晚期早产儿(孕36周),其新生儿期起病的间质性肺病最终被诊断为MPS I。该新生儿需要长期呼吸支持和氧疗,这进一步增加了遗传性肺表面活性物质功能障碍的诊断可能性。全外显子测序在观察到α-L-艾杜糖醛酸酶水平较低后确诊为MPS I。结果强调了对于持续呼吸功能不全的新生儿,有必要考虑MPS I相关的肺部受累情况。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/af8e/10204566/569fa3e725c0/diseases-11-00067-g001.jpg

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