Literature DB >> 32331969

α-Mannosidosis - An underdiagnosed lysosomal storage disease in individuals with an 'MPS-like' phenotype.

Thomas Wiesinger1, Markus Schwarz2, Thomas P Mechtler2, Sandra Liebmann-Reindl2, Berthold Streubel3, David C Kasper4.   

Abstract

Individuals affected by alpha-Mannosidosis suffer from similar clinical symptoms such as respiratory infections, skeletal changes as patients with mucopolysaccharidoses (MPS). α-Mannosidosis is considered as an ultra-rare disorders and also diagnostic testing is often limited. With the availability of novel therapies and easy-to-access diagnostic tests (e.g. Tandem mass spectrometry) using dried blood spots for both enzymatic and genetic testing, the chance for the development of a better understanding of disease and awareness may be triggered. In a pilot study, we have investigated 1010 residual dried blood spot samples from individuals suspicious to MPS. In these study cohort, 158/1010 individuals were genetically confirmed for MPS. Additional biochemical and genetic confirmatory testing for α-mannosidases revealed four individuals with a final diagnosis of α-mannosidosis. This unexpected high number of individuals with α-mannosidosis demonstrated the urgent need of taking this rare disorder in clinical and diagnostic consideration particularly in patients suspicious to MPS.
Copyright © 2020 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  Dried blood spots; Lysosomal storage disease; Mucopolysaccharidosis; Rare disease; Tandem mass spectrometry; α-Mannosidosis; α-mannosidase

Mesh:

Substances:

Year:  2020        PMID: 32331969     DOI: 10.1016/j.ymgme.2020.04.001

Source DB:  PubMed          Journal:  Mol Genet Metab        ISSN: 1096-7192            Impact factor:   4.797


  4 in total

1.  Long-term outcome of patients with alpha-mannosidosis - A single center study.

Authors:  Patryk Lipiński; Agnieszka Różdżyńska-Świątkowska; Katarzyna Iwanicka-Pronicka; Barbara Perkowska; Paulina Pokora; Anna Tylki-Szymańska
Journal:  Mol Genet Metab Rep       Date:  2021-12-09

Review 2.  Mortality in patients with alpha-mannosidosis: a review of patients' data and the literature.

Authors:  Julia B Hennermann; Eva M Raebel; Francesca Donà; Marie-Line Jacquemont; Graziella Cefalo; Andrea Ballabeni; Dag Malm
Journal:  Orphanet J Rare Dis       Date:  2022-07-23       Impact factor: 4.303

3.  Twenty years of Colombian experience with enzymatic screening in patients with features of mucopolysaccharidosis.

Authors:  Alfredo Uribe-Ardila; Johana Ramirez-Borda; Adis Ayala
Journal:  JIMD Rep       Date:  2022-07-28

4.  The SPARKLE registry: protocol for an international prospective cohort study in patients with alpha-mannosidosis.

Authors:  Julia B Hennermann; Nathalie Guffon; Federica Cattaneo; Ferdinando Ceravolo; Line Borgwardt; Allan M Lund; Mercedes Gil-Campos; Anna Tylki-Szymanska; Nicole M Muschol
Journal:  Orphanet J Rare Dis       Date:  2020-09-29       Impact factor: 4.123

  4 in total

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