Literature DB >> 21783023

Neuroimaging findings in a brain with Niemann-Pick type C disease.

Jei-Yie Huang1, Sing-Fung Peng, Chih-Chao Yang, Kuo-Yang Yen, Kai-Yuan Tzen, Ruoh-Fang Yen.   

Abstract

Niemann-Pick type C disease (NPC) is a rare autosomal recessive lipid storage disorder caused by impaired cellular functions in processing and transporting low-density lipoprotein-cholesterol. In this report, we present magnetic resonance imaging (MRI), magnetic resonance spectrography (MRS) and 18-fluoro-2-deoxyglucose positron emission tomography (PET) imaging results for a 22-year-old male NPC patient. The patient's two MRI studies (at age 19 years and 22 years) demonstrated progressive changes of brain atrophy that were more prominent at the frontal lobes, and hyperintense signals in bilateral parietal-occipital periventricular white matter. MRS (at age 19 years) revealed no significant decrease in N-acetyl aspartate/choline ratio in the left frontal central white matter. PET (at age 22 years) showed significant bilateral hypometabolism in the prefrontal cortex and dorsomedial thalamus, and hypermetabolism in the parietal-occipital white matter, lenticular nucleus of the basal ganglia, cerebellum and pons. The imaging findings noted by MRI, MRS and 18-fluoro-2-deoxyglucose PET offered a possible supplementary explanation for the clinical neurological symptoms of this NPC patient.
Copyright © 2011 Formosan Medical Association & Elsevier. Published by Elsevier B.V. All rights reserved.

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Year:  2011        PMID: 21783023     DOI: 10.1016/S0929-6646(11)60080-6

Source DB:  PubMed          Journal:  J Formos Med Assoc        ISSN: 0929-6646            Impact factor:   3.282


  15 in total

1.  Heterogeneity and frequency of movement disorders in juvenile and adult-onset Niemann-Pick C disease.

Authors:  Mathieu Anheim; Ouhaïd Lagha-Boukbiza; Marie-Céline Fleury-Lesaunier; Maria-Paola Valenti-Hirsch; Edouard Hirsch; Hélène Gervais-Bernard; Emmanuel Broussolle; Stéphane Thobois; Marie T Vanier; Philippe Latour; Christine Tranchant
Journal:  J Neurol       Date:  2013-11-01       Impact factor: 4.849

Review 2.  Do heterozygous mutations of Niemann-Pick type C predispose to late-onset neurodegeneration: a review of the literature.

Authors:  Susanne A Schneider; Sabina Tahirovic; John Hardy; Michael Strupp; Tatiana Bremova-Ertl
Journal:  J Neurol       Date:  2019-11-07       Impact factor: 4.849

3.  Phenotypic heterogeneity of Niemann-Pick disease type C in monozygotic twins.

Authors:  Alberto Benussi; Antonella Alberici; Enrico Premi; Valeria Bertasi; Maria Sofia Cotelli; Marinella Turla; Andrea Dardis; Stefania Zampieri; Eleonora Marchina; Barbara Paghera; Francesca Gallivanone; Isabella Castiglioni; Alessandro Padovani; Barbara Borroni
Journal:  J Neurol       Date:  2014-12-24       Impact factor: 4.849

Review 4.  Lysosomal Leukodystrophies Lysosomal Storage Diseases Associated With White Matter Abnormalities.

Authors:  Gustavo H B Maegawa
Journal:  J Child Neurol       Date:  2019-02-13       Impact factor: 1.987

5.  Vestibular function in patients with Niemann-Pick type C disease.

Authors:  Tatiana Bremova; Siegbert Krafczyk; Stanislavs Bardins; Jörg Reinke; Michael Strupp
Journal:  J Neurol       Date:  2016-08-20       Impact factor: 4.849

6.  Brain hypometabolism in rare genetic neurodegenerative disease: Niemann-Pick disease type C, spinocerebellar ataxia and Huntington disease assessed by FDG PET.

Authors:  Yung Hsiang Kao; Melissa Cheng; Dennis Velakoulis; Mark Walterfang; Dinesh Sivaratnam
Journal:  Asia Ocean J Nucl Med Biol       Date:  2021

7.  Early glial activation, synaptic changes and axonal pathology in the thalamocortical system of Niemann-Pick type C1 mice.

Authors:  Sarah N R Pressey; David A Smith; Andrew M S Wong; Frances M Platt; Jonathan D Cooper
Journal:  Neurobiol Dis       Date:  2011-12-16       Impact factor: 5.996

Review 8.  Recent neuroimaging, neurophysiological, and neuropathological advances for the understanding of NPC.

Authors:  Alberto Benussi; Maria Sofia Cotelli; Alessandro Padovani; Barbara Borroni
Journal:  F1000Res       Date:  2018-02-15

9.  Quantitative magnetic resonance imaging of brain atrophy in a mouse model of Niemann-Pick type C disease.

Authors:  John W Totenhagen; Adam Bernstein; Eriko S Yoshimaru; Robert P Erickson; Theodore P Trouard
Journal:  PLoS One       Date:  2017-05-24       Impact factor: 3.240

10.  Pre-symptomatic activation of antioxidant responses and alterations in glucose and pyruvate metabolism in Niemann-Pick Type C1-deficient murine brain.

Authors:  Barry E Kennedy; Veronique G LeBlanc; Tiffany M Mailman; Debra Fice; Ian Burton; Tobias K Karakach; Barbara Karten
Journal:  PLoS One       Date:  2013-12-18       Impact factor: 3.240

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