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DOI 10.1016/j.nmd.2021.02.013

Медицинская библиотека · Neuromuscular disorders : NMD · 2021

Late onset Pompe Disease in India - Beyond the Caucasian phenotype.

Puri Ratna Dua, Setia Nitika, N Vinu, Jagadeesh Sujatha, Nampoothiri Sheela, Gupta Neerja, Muranjan Mamta, Bhat Meenakshi, Girisha Katta M, Kabra Madhulika, Verma Jyotsna, Thomas Divya C, Biji Ishpreet, Raja Jayarekha, Makkar Ravinder, Verma Ishwar C, Kishnani Priya S

СтатьяМногоцентровое исследованиеПоддержка исследования: вне правительства СШАПеревод в очереди

Аннотация

We evaluated the clinical histories, motor and pulmonary functions, cardiac phenotypes and GAA genotypes of an Indian cohort of twenty patients with late onset Pompe disease (LOPD) in this multi-centre study. A mean age at onset of symptoms and diagnosis of 9.9 ± 9.7 years and 15.8 ± 12.1 years respectively was identified. All patients had lower extremity limb-girdle muscle weakness. Seven required ventilatory support and seven used mobility assists. Of the four who used both assists, two received ventilatory support prior to wheelchair use. Cardiac involvement was seen in eight patients with various combinations of left ventricular hypertrophy, tricuspid regurgitation, cardiomyopathy, dilated ventricles with biventricular dysfunction and aortic regurgitation. Amongst 20 biochemically diagnosed patients (low residual GAA enzyme activity) GAA genotypes of 19 patients identified homozygous variants in eight and compound heterozygous in 11: 27 missense, 3 nonsense, 2 initiator codon, 3 splice site and one deletion. Nine variants in 7 patients were novel. The leaky Caucasian, splice site LOPD variant, c.-32-13T>G mutation was absent. This first study from India provides an insight into a more severe LOPD phenotype with earlier disease onset at 9.9 years compared to 33.3 years in Caucasian patients, and cardiac involvement more than previously reported. The need for improvement in awareness and diagnosis of LOPD in India is highlighted.

Для цитирования: Puri Ratna Dua, Setia Nitika, N Vinu, Jagadeesh Sujatha, Nampoothiri Sheela, Gupta Neerja, Muranjan Mamta, Bhat Meenakshi, Girisha Katta M, Kabra Madhulika, Verma Jyotsna, Thomas Divya C, Biji Ishpreet, Raja Jayarekha, Makkar Ravinder, Verma Ishwar C, Kishnani Priya S. Late onset Pompe Disease in India - Beyond the Caucasian phenotype.. Neuromuscular disorders : NMD. 2021. DOI: 10.1016/j.nmd.2021.02.013.

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