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Updated: Sep 11, 2025

In Vitro Enzyme Measurement to Test Pharmacological Chaperone Responsiveness in Fabry and Pompe Disease
Published on: December 20, 2017
Latest Advancements in Treatment Options for Infantile-Onset Pompe Disease: A Comprehensive Narrative Review
Misha Khan1, Mufliha Ibrahim1, Farhana Riaz1
1Department of Medicine and Surgery, Liaquat National Hospital and Medical College, Karachi, Pakistan.
Insights
Infantile-onset Pompe disease (IOPD) is a severe genetic disorder. Enzyme replacement therapy improves survival, but new strategies are needed to overcome its limitations and enhance treatment for better patient outcomes.
Area of Science:
- Biochemistry
- Genetics
- Pediatrics
Background:
- Infantile-onset Pompe disease (IOPD) is a rare genetic disorder caused by acid alpha-glucosidase deficiency.
- Glycogen accumulation in organs leads to severe complications like muscle weakness and cardiomyopathy, with high mortality without treatment.
Purpose of the Study:
- To review current therapeutic strategies for IOPD.
- To examine the efficacy, challenges, and future directions of IOPD treatments.
Main Methods:
- Literature review of current and emerging therapies for IOPD.
- Analysis of enzyme replacement therapy (ERT) efficacy and limitations.
- Exploration of adjunctive therapies and diagnostic advancements.
Main Results:
- Enzyme replacement therapy (ERT) with recombinant human GAA is the standard treatment, improving survival and cardiac outcomes.
- ERT limitations include immune responses, poor muscle uptake, and inability to cross the blood-brain barrier.
- Emerging therapies focus on enhancing ERT, gene therapy, and immune modulation.
Conclusions:
- Early diagnosis and timely intervention, including newborn screening, are critical for better IOPD outcomes.
- Advancements in adjunctive therapies show promise in overcoming ERT limitations.
- Future research directions aim to improve enzyme delivery, function, and overall treatment efficacy for IOPD patients.
Abstract:
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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