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DOI 10.1002/mus.21291

Медицинская библиотека · Muscle & nerve · 2009

A novel mutation of the GAA gene in a Finnish late-onset Pompe disease patient: clinical phenotype and follow-up with enzyme replacement therapy.

Korpela Mari P, Paetau Anders, Löfberg Mervi I, Timonen Marjut H, Lamminen Antti E, Kiuru-Enari Sari M K

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Аннотация

Pompe disease is a rare, progressive disease leading to skeletal muscle weakness due to deficiency of the acid alpha-glucosidase (GAA) enzyme. Herein we report the first diagnosed Finnish patient with a phenotype compatible with the late-onset form of Pompe disease. Molecular genetic analysis of the GAA gene revealed a novel missense mutation, 1725C>A (Y575X), combined with a previously reported mutation, 1634C>T (P545L). Human recombinant alpha-glucosidase enzyme (alglucosidase-alpha) treatment was initiated for this patient at age 20 years. After 12 months she was no longer fully wheelchair-bound, and muscle strength had improved. No disease progression was visible on muscle magnetic resonance imaging of the lower limbs, and the energy state of the muscle cells increased by 46% on phosphorus magnetic resonance spectroscopy. Overall, our findings suggest that enzyme replacement therapy is indicated, even in patients with late-onset Pompe disease, to halt disease progression and improve the quality of daily life.

Для цитирования: Korpela Mari P, Paetau Anders, Löfberg Mervi I, Timonen Marjut H, Lamminen Antti E, Kiuru-Enari Sari M K. A novel mutation of the GAA gene in a Finnish late-onset Pompe disease patient: clinical phenotype and follow-up with enzyme replacement therapy.. Muscle & nerve. 2009. DOI: 10.1002/mus.21291.

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