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DOI 10.1016/j.ymgmr.2023.101014

Медицинская библиотека · Molecular genetics and metabolism reports · 2023

The diagnostic journey for patients with late-onset GM2 Gangliosidoses.

Lopshire Mariah C, Tifft Cynthia, Burns John, Gould Rebecca, Zheng Riliang, Batsu Isabela

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Аннотация

Late-onset forms of GM2 gangliosidosis-mainly, Tay-Sachs disease and Sandhoff disease-are under-recognized in clinical practice. In these rare lysosomal storage disorders, deficiency of β-hexosaminidase A results in excessive accumulation of GM2 ganglioside primarily within neurons, leading to cell death and progressive neurodegenerative symptoms, including ataxia, dysarthria, muscle weakness, tremors, atrophy, and psychosis. Presentation is variable and often mimics more common neurodegenerative disorders. We conducted semi-structured interviews on GM2 gangliosidoses diagnosis and treatment with five experts, 30 neurologists, and 28 patients and caregivers. Symptom onset occurred during adolescence/early adulthood in 92% of patients (median age: 14 years). Patients first visited a healthcare provider at a median age of 20 years and received a GM2 diagnosis at a median age of 26 years. Nearly all patients reported problems with their legs and balance starting from symptom onset. Problems with memory, attention span, speech and fatigue were reported more after diagnosis. Patients visited an average of eight healthcare providers before receiving a diagnosis; 64% were diagnosed by a neurologist. Four neurologists (13%) in our sample were aware that there are late-onset forms of GM2 gangliosidosis. The path to diagnosis is long for this late-onset form of a classically fatal infantile disease.

Для цитирования: Lopshire Mariah C, Tifft Cynthia, Burns John, Gould Rebecca, Zheng Riliang, Batsu Isabela. The diagnostic journey for patients with late-onset GM2 Gangliosidoses.. Molecular genetics and metabolism reports. 2023. DOI: 10.1016/j.ymgmr.2023.101014.
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