Literature DB >> 21466370

Mucolipidosis type III α/β: the first characterization of this rare disease by autopsy.

Darcy A Kerr1, Vincent A Memoli, Sara S Cathey, Brent T Harris.   

Abstract

We report findings from an autopsy of a 45-year-old woman with the rare lysosomal storage disease mucolipidosis type III α/β. Her disease manifested most notably as multiple bone and cartilage problems with tracheal and bronchial malacia. Principal autopsy findings included gross abnormalities in bone and cartilage with corresponding microscopic cytoplasmic lysosomal granules. These cytoplasmic granules were also seen in histologic preparations of the brain, myocardium, heart valves, and fibroblasts of the liver and skin by light and electron microscopy. By electron microscopy there were scattered, diffuse vesicular cytoplasmic granules in neurons and glia and an increase in lysosomal structures with fine electron lucent granularity in the above tissue types. Our findings help elaborate current understanding of this disease and differentiate it from the mucopolysaccharidoses and related disorders. To our knowledge, this is the first report to document pathologic findings in a patient with mucolipidosis type III α/β by autopsy.

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Year:  2011        PMID: 21466370      PMCID: PMC4188553          DOI: 10.5858/2010-0236-CR.1

Source DB:  PubMed          Journal:  Arch Pathol Lab Med        ISSN: 0003-9985            Impact factor:   5.534


  16 in total

1.  Genomic organisation of the UDP-N-acetylglucosamine-1-phosphotransferase gamma subunit (GNPTAG) and its mutations in mucolipidosis III.

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Review 2.  Human mucopolysaccharidosis IIID: clinical, biochemical, morphological and immunohistochemical characteristics.

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Journal:  J Neuropathol Exp Neurol       Date:  1997-10       Impact factor: 3.685

3.  I-cell disease (mucolipidosis II): a lysosomopathy.

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Journal:  Pediatrics       Date:  1974-12       Impact factor: 7.124

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Journal:  J Inherit Metab Dis       Date:  2002-12       Impact factor: 4.982

6.  Cardiac involvement in mucolipidosis. Importance of non-invasive studies for detection of cardiac abnormalities.

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Journal:  J Rheumatol       Date:  1993-01       Impact factor: 4.666

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Journal:  Hum Pathol       Date:  1994-03       Impact factor: 3.466

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Journal:  Pediatr Radiol       Date:  1985

10.  Mucolipidosis II (I-cell disease) and mucolipidosis IIIA (classical pseudo-hurler polydystrophy) are caused by mutations in the GlcNAc-phosphotransferase alpha / beta -subunits precursor gene.

Authors:  Mariko Kudo; Michael S Brem; William M Canfield
Journal:  Am J Hum Genet       Date:  2006-01-24       Impact factor: 11.025

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  5 in total

1.  Distinct Modes of Balancing Glomerular Cell Proteostasis in Mucolipidosis Type II and III Prevent Proteinuria.

Authors:  Wiebke Sachs; Marlies Sachs; Elke Krüger; Stephanie Zielinski; Oliver Kretz; Tobias B Huber; Anke Baranowsky; Lena Marie Westermann; Renata Voltolini Velho; Nataniel Floriano Ludwig; Timur Alexander Yorgan; Giorgia Di Lorenzo; Katrin Kollmann; Thomas Braulke; Ida Vanessa Schwartz; Thorsten Schinke; Tatyana Danyukova; Sandra Pohl; Catherine Meyer-Schwesinger
Journal:  J Am Soc Nephrol       Date:  2020-07-08       Impact factor: 10.121

2.  Next Generation Sequencing identifies mutations in GNPTG gene as a cause of familial form of scleroderma-like disease.

Authors:  Abdelali Zrhidri; Saadia Amasdl; Jaber Lyahyai; Hanane Elouardi; Bouchra Chkirate; Laure Raymond; Grégory Egéa; Mohamed Taoudi; Said El Mouatassim; Abdelaziz Sefiani
Journal:  Pediatr Rheumatol Online J       Date:  2017-09-26       Impact factor: 3.054

3.  Mucolipidosis type III, a series of adult patients.

Authors:  Esmee Oussoren; David van Eerd; Elaine Murphy; Robin Lachmann; Jan C van der Meijden; Lies H Hoefsloot; Rob Verdijk; George J G Ruijter; Mario Maas; Carla E M Hollak; Janneke G Langendonk; Ans T van der Ploeg; Mirjam Langeveld
Journal:  J Inherit Metab Dis       Date:  2018-04-27       Impact factor: 4.982

Review 4.  Lysosome function in glomerular health and disease.

Authors:  Catherine Meyer-Schwesinger
Journal:  Cell Tissue Res       Date:  2021-01-12       Impact factor: 4.051

5.  Case Report: Mucolipidosis II and III Alpha/Beta Caused by Pathogenic Variants in the GNPTAB Gene (Mucolipidosis).

Authors:  Shao-Jia Mao; Yu-Mei Zu; Yang-Li Dai; Chao-Chun Zou
Journal:  Front Pediatr       Date:  2022-04-08       Impact factor: 3.418

  5 in total

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