Literature DB >> 17182441

Skin biopsy: a useful tool in the diagnosis of lysosomal storage diseases.

Joseph Alroy1, Angelo A Ucci.   

Abstract

In this report, the authors summarize their 19-year experience with over 200 biochemically proven cases of lysosomal storage diseases using electron microscopic screening of more than 950 skin biopsies. They found that electron microscopy (EM) is a highly sensitive, efficient, cost-effective, and rapid diagnostic screening tool for evaluation of lysosomal storage diseases in skin biopsies. Although EM is more expensive than a single enzyme assay, it can exclude more than 90% of cases in which lysosomal storage disease is being considered. EM is critical for diagnosis of neuronal ceroid lipofuscinosis and mucolipidosis IV and is the most cost-effective screening tool in patients with previously unrecognized storage diseases.

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Year:  2006        PMID: 17182441     DOI: 10.1080/01913120500520986

Source DB:  PubMed          Journal:  Ultrastruct Pathol        ISSN: 0191-3123            Impact factor:   1.094


  7 in total

Review 1.  [Lysosomal storage diseases: A brief summary].

Authors:  A Bornemann; Klaus Harzer
Journal:  Pathologe       Date:  2015-09       Impact factor: 1.011

2.  The use of electron microscopy for diagnosis of malignant pleural mesothelioma.

Authors:  Oscar Arrieta; Zyanya Lucia Zatarain-Barron; Amir Carmona; Hugo Domínguez-Malagón
Journal:  J Thorac Dis       Date:  2017-03       Impact factor: 2.895

3.  Lessons learnt from animal models: pathophysiology of neuropathic lysosomal storage disorders.

Authors:  Kim M Hemsley; John J Hopwood
Journal:  J Inherit Metab Dis       Date:  2010-05-07       Impact factor: 4.982

Review 4.  Mucolipidosis type IV: an update.

Authors:  Kazuyo Wakabayashi; Ann Marie Gustafson; Ellen Sidransky; Ehud Goldin
Journal:  Mol Genet Metab       Date:  2011-06-16       Impact factor: 4.797

5.  LAPTMs regulate lysosomal function and interact with mucolipin 1: new clues for understanding mucolipidosis type IV.

Authors:  Silvia Vergarajauregui; Jose A Martina; Rosa Puertollano
Journal:  J Cell Sci       Date:  2011-01-11       Impact factor: 5.285

6.  Shared functional defect in IP₃R-mediated calcium signaling in diverse monogenic autism syndromes.

Authors:  G Schmunk; B J Boubion; I F Smith; I Parker; J J Gargus
Journal:  Transl Psychiatry       Date:  2015-09-22       Impact factor: 6.222

7.  A novel role for 12/15-lipoxygenase in regulating autophagy.

Authors:  Alwena H Morgan; Victoria J Hammond; Machiko Sakoh-Nakatogawa; Yoshinori Ohsumi; Christopher P Thomas; Fabien Blanchet; Vincent Piguet; Kirill Kiselyov; Valerie B O'Donnell
Journal:  Redox Biol       Date:  2014-12-03       Impact factor: 11.799

  7 in total

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