婴儿起病庞贝病治疗方案的最新进展:一项全面的叙述综述。

IF 1.2 Q2 PEDIATRICS
Clinical Medicine Insights-Pediatrics Pub Date : 2025-08-11 eCollection Date: 2025-01-01 DOI:10.1177/11795565251337900
Misha Khan, Mufliha Ibrahim, Farhana Riaz, Zainab Awan, Eeshal Zulfiqar, Akash Kumar, Ahmed Sermed Al Sakini, Tariq Mahmood Khan, Ali Saad Al-Shammari
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引用次数: 0

摘要

婴儿起病庞贝病(IOPD)是一种罕见的遗传性疾病,与溶酶体酶,酸性α -葡萄糖苷酶的缺乏有关。其特点是溶酶体和非溶酶体结合的糖原在心脏、骨骼肌和脑组织等各器官中积累,导致肌肉无力、肥厚性心肌病、呼吸功能不全等并发症。如果不进行治疗,IOPD的死亡率非常高,患者在生命的第一年就会死亡。在过去的几十年里,在改善IOPD患者的预后和生活质量方面取得了显著的治疗进展。酶替代疗法(ERT)与重组人GAA已成为治疗的基石,证明有效延长生存和减少心脏肥厚。然而,ERT有局限性,包括免疫反应的发展,骨骼肌摄取不一致,以及无法穿过脑屏障。最近的研究集中在通过辅助治疗如免疫调节、基因治疗和伴侣介导的方法来改善酶的传递和功能来增强ERT。此外,早期诊断(包括新生儿筛查)方面的进步使及时干预成为可能,这对取得更好的结果至关重要。这篇综述全面检查了目前的治疗策略,它们的疗效,挑战和未来的方向管理这种疾病。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Latest Advancements in Treatment Options for Infantile-Onset Pompe Disease: A Comprehensive Narrative Review.

Infantile-onset Pompe disease (IOPD) is a rare genetic disorder associated with a deficiency of a lysosomal enzyme, the acid alpha-glucosidase. It is characterized by the accumulation of lysosomal and non-lysosomal-bound glycogen in various organs, such as the heart, skeletal muscle, and brain tissue, resulting in muscle weakness, hypertrophic cardiomyopathy, respiratory insufficiency, and other complications. Without treatment, IOPD has a very high mortality rate, with patients dying within the first year of life. Over the past few decades, significant therapeutic advancements have been made to improve the prognosis and quality of life for IOPD patients. Enzyme replacement therapy (ERT) with recombinant human GAA has been the cornerstone of treatment, demonstrating efficacy in prolonging survival and reducing cardiac hypertrophy. However, ERT has limitations, including the development of immune responses, inconsistent skeletal muscle uptake, and the inability to cross the brain barrier. Recent research has focused on enhancing ERT through adjunctive therapies such as immune modulation, gene therapy, and chaperone-mediated approaches to improve enzyme delivery and function. Additionally, advancements in early diagnosis, including newborn screening, have enabled timely intervention, which is crucial for better outcomes. This review comprehensively examines the current therapeutic strategies for IOPD, their efficacy, challenges, and future directions for managing this disease.

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