Literature DB >> 23906593

Niemann-Pick type C: a potentially treatable disorder?

Ammar Kheder1, Camilla Scott, Simon Olpin, Marios Hadjivassiliou.   

Abstract

Niemann-Pick disease refers to a group of autosomal recessive lipid storage disorders associated with a variable degree of neurological manifestations in addition to other organ involvement. Niemann-Pick disease is divided into types A-C. Of interest to neurologists is Niemann-Pick type C because of the association with neurological manifestations that are not confined to childhood. The clinical presentation of Niemann-Pick type C is variable, depending on age at onset. Neurological symptoms vary with age: hypotonia, delay in developmental motor milestones, falls, seizures, learning difficulties, ataxia with cognitive deficits and psychosis. The definitive diagnosis of Niemann-Pick type C requires demonstration of abnormal intracellular cholesterol trafficking using the filipin test. Therapeutic interventions are few but one that is of interest is miglustat, which has been approved for specific treatment of the neurological manifestations. It showed improvement in horizontal saccadic eye movement and a trend towards improvement or stabilisation in swallowing, hearing and walking. Niemann-Pick type C should be considered in patients with early-onset ataxia associated with progressive learning/cognitive difficulties even in the absence of vertical gaze palsy.

Entities:  

Keywords:  BEHAVIOURAL DISORDER; CEREBELLAR ATAXIA; COGNITION; DEMENTIA; DYSTONIA

Mesh:

Substances:

Year:  2013        PMID: 23906593     DOI: 10.1136/practneurol-2013-000525

Source DB:  PubMed          Journal:  Pract Neurol        ISSN: 1474-7758


  5 in total

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Authors:  Kavitha Siva; Giuseppina Covello; Michela A Denti
Journal:  Nucleic Acid Ther       Date:  2014-02       Impact factor: 5.486

Review 2.  Movement Disorders and Hematologic Diseases.

Authors:  Roshni Abee Patel; Deborah A Hall; Sheila Eichenseer; Meagan Bailey
Journal:  Mov Disord Clin Pract       Date:  2020-12-29

Review 3.  The genetics of ataxia: through the labyrinth of the Minotaur, looking for Ariadne's thread.

Authors:  M Mancuso; D Orsucci; G Siciliano; U Bonuccelli
Journal:  J Neurol       Date:  2014-09       Impact factor: 4.849

Review 4.  Consensus clinical management guidelines for Niemann-Pick disease type C.

Authors:  Tarekegn Geberhiwot; Alessandro Moro; Andrea Dardis; Uma Ramaswami; Sandra Sirrs; Mercedes Pineda Marfa; Marie T Vanier; Mark Walterfang; Shaun Bolton; Charlotte Dawson; Bénédicte Héron; Miriam Stampfer; Jackie Imrie; Christian Hendriksz; Paul Gissen; Ellen Crushell; Maria J Coll; Yann Nadjar; Hans Klünemann; Eugen Mengel; Martin Hrebicek; Simon A Jones; Daniel Ory; Bruno Bembi; Marc Patterson
Journal:  Orphanet J Rare Dis       Date:  2018-04-06       Impact factor: 4.123

5.  Niemann-Pick type C: contemporary diagnosis and treatment of a classical disorder.

Authors:  Meher Lad; Rhys H Thomas; Kirstie Anderson; Timothy D Griffiths
Journal:  Pract Neurol       Date:  2019-06-26
  5 in total

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