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DOI 10.1177/0883073812464526

Медицинская библиотека · Journal of child neurology · 2012

Effects of miglustat on stabilization of neurological disorder in niemann-pick disease type C: Iranian pediatric case series.

Karimzadeh Parvaneh, Tonekaboni Seyed Hassan, Ashrafi Mahmoud Reza, Shafeghati Yousef, Rezayi Alireza, Salehpour Shadab, Ghofrani Mohammad, Taghdiri Mohammad Mehdi, Rahmanifar Ali, Zaman Talieh, Aryani Omid, Shoar Babak Najaf, Shiva Farideh, Tavasoli Alireza, Houshmand Massoud

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Аннотация

Niemann-Pick disease type C is a rare neurodegenerative disorder with autosomal recessive inheritance that can be broadly categorized into different forms dependent on age at disease onset: pre-/perinatal, early infantile, late infantile, juvenile, and adolescent/adult. This study was conducted to define the age at onset, clinical manifestations, neuroimaging findings and response to treatment in 21 patients diagnosed with Niemann-Pick disease type C and managed in the neurology departments of hospitals in Tehran, Iran. The effects of miglustat on patient ambulation, fine and gross motor function, swallowing, hearing, speech, seizures, psychomotor development, and ocular movements were evaluated for up to 26 months of treatment. Ambulation, fine and gross motor movements, swallowing, speech, and supranuclear gaze palsy were generally stabilized during therapy, and psychomotor delay appeared to be improved in early- and late-infantile onset patients. However, miglustat had no effect on organomegaly or other systemic manifestations of the disease. Miglustat was well tolerated.

Для цитирования: Karimzadeh Parvaneh, Tonekaboni Seyed Hassan, Ashrafi Mahmoud Reza, Shafeghati Yousef, Rezayi Alireza, Salehpour Shadab, Ghofrani Mohammad, Taghdiri Mohammad Mehdi, Rahmanifar Ali, Zaman Talieh, Aryani Omid, Shoar Babak Najaf, Shiva Farideh, Tavasoli Alireza, Houshmand Massoud. Effects of miglustat on stabilization of neurological disorder in niemann-pick disease type C: Iranian pediatric case series.. Journal of child neurology. 2012. DOI: 10.1177/0883073812464526.

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