Literature DB >> 10967652

[An autopsy case of juvenile neuronal ceroid-lipofuscinosis with dilated cardiomyopathy].

H Tomiyasu1, W Takahashi, T Ohta, F Yoshii, M Shibuya, Y Shinohara.   

Abstract

We reported an autopsy case of neuronal ceroid-lipofuscinosis (NCL3) with dilatated cardiomyopathy. A 29-year-old male patient first noticed night-blindness at the age of four years. He was pointed out retinitis pigmentosa at the age of six years and developed ataxia, mental retardation, epilepsy and myoclonus, thereafter. T1 weighted MRI showed diffuse atrophy of the cerebellum, brainstem, and cerebrum, and dilatation of the ventricular system and T2-weighted MRI showed mild high signal intensity in the white matter around the trigones of the lateral ventricles. Autopsy findings showed an abundant accumulation of ceroid-lipofuscin-like lipopigments in most neurons in the central nervous system, and curvilinear bodies and lipofuscin like granules were confirmed by electron microscopy. The heart muscle showed an increase in the accumulation of ceroid-lipofuscin-like lipopigments, severe fibrosis and fatty infiltration in the myocardium. The peculiar point of this case is NCL3 with dilated cardiomyopathy.

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Year:  2000        PMID: 10967652

Source DB:  PubMed          Journal:  Rinsho Shinkeigaku        ISSN: 0009-918X


  4 in total

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Authors:  Anne-Hélène Lebrun; Parisa Moll-Khosrawi; Sandra Pohl; Georgia Makrypidi; Stephan Storch; Dirk Kilian; Thomas Streichert; Benjamin Otto; Sara E Mole; Kurt Ullrich; Susan Cotman; Alfried Kohlschütter; Thomas Braulke; Angela Schulz
Journal:  Mol Med       Date:  2011-08-18       Impact factor: 6.354

2.  A murine model of infantile neuronal ceroid lipofuscinosis-ultrastructural evaluation of storage in the central nervous system and viscera.

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3.  Cathepsin D deficiency causes juvenile-onset ataxia and distinctive muscle pathology.

Authors:  Joshua Hersheson; Derek Burke; Robert Clayton; Glenn Anderson; Thomas S Jacques; Philippa Mills; Nicholas W Wood; Paul Gissen; Peter Clayton; Julian Fearnley; Sara E Mole; Henry Houlden
Journal:  Neurology       Date:  2014-10-08       Impact factor: 9.910

4.  Tripeptidyl Peptidase 1 (TPP1) Deficiency in a 36-Year-Old Patient with Cerebellar-Extrapyramidal Syndrome and Dilated Cardiomyopathy.

Authors:  Agnieszka Ługowska; Joanna K Purzycka-Olewiecka; Rafał Płoski; Grażyna Truszkowska; Maciej Pronicki; Paulina Felczak; Mateusz Śpiewak; Aleksandra Podlecka-Piętowska; Martyna Sitek; Zofia T Bilińska; Przemysław Leszek; Małgorzata Bednarska-Makaruk
Journal:  Life (Basel)       Date:  2021-12-21
  4 in total

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