Pediatric neurology · 2017
Management Strategies for CLN2 Disease.
СтатьяОбзорНазвание и аннотация: Перевод пока не подготовлен
Обработка статьи · этапы, ошибки и управление
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Ошибки и ограничения
История · было → стало
Состояние обновляется каждые 5 секунд, пока блок открыт. Процент показывается только там, где известен объём работы. Пауза останавливает продолжение этапа; текущий запрос к источнику может завершиться.
Williams Ruth E, Adams Heather R, Blohm Martin, Cohen-Pfeffer Jessica L, de Los Reyes Emily, Denecke Jonas, Drago Kristen, Fairhurst Charlie, Frazier Margie, Guelbert Norberto, Kiss Szilárd, Kofler Annamaria, Lawson John A, Lehwald Lenora, Leung Mary-Anne, Mikhaylova Svetlana, Mink Jonathan W, Nickel Miriam, Shediac Renée, Sims Katherine, Specchio Nicola, Topcu Meral, von Löbbecke Ina, West Andrea, Zernikow Boris, Schulz Angela
Аннотация
CLN2 disease (neuronal ceroid lipofuscinosis type 2) is a rare, autosomal recessive, pediatric-onset, rapidly progressive neurodegenerative lysosomal storage disorder caused by tripeptidyl peptidase 1 (TPP1) enzyme deficiency, and is characterized by language delay, seizures, rapid cognitive and motor decline, blindness, and early death. No management guidelines exist and there is a paucity of published disease-specific evidence to inform clinical practice, which currently draws upon experience from the field of childhood neurodisability. Twenty-four disease experts were surveyed on CLN2 disease management and a subset met to discuss current practice. Management goals and strategies are consistent among experts globally and are guided by the principles of pediatric palliative care. Goals and interventions evolve as the disease progresses, with a shift in focus from maintenance of function early in the disease to maintenance of quality of life. A multidisciplinary approach is critical for optimal patient care. This work represents an initial step toward the development of consensus-based management guidelines for CLN2 disease.