Literature DB >> 23307885

Alpha-mannosidosis: a report of 2 siblings and review of the literature.

Rajeshree Govender1, Lawrence Mubaiwa.   

Abstract

Alpha-mannosidosis is a rare lysosomal storage disorder with a heterogeneous clinical presentation. We describe a set of siblings with alpha-mannosidosis. The older child presented with a severe phenotype with multisystem involvement and had progressive deterioration in her motor and cognitive functioning. She had a poor outcome. The second child has a less severe disease course. He is being managed symptomatically only because of the family's poor socioeconomic circumstances. Therapeutic options in this condition are limited to bone marrow transplant early in the disease course. These disorders, although rare, should be considered in the approach to a child with dysmorphism, developmental delay, skeletal deformities, and visceromegaly.

Entities:  

Keywords:  alpha-mannosidosis; dysmorphic; hepatosplenomegaly; lysosomal storage disorders

Mesh:

Substances:

Year:  2013        PMID: 23307885     DOI: 10.1177/0883073812470973

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  5 in total

1.  Cognitive profile and activities of daily living: 35 patients with alpha-mannosidosis.

Authors:  L Borgwardt; A M Thuesen; K J Olsen; J Fogh; C I Dali; A M Lund
Journal:  J Inherit Metab Dis       Date:  2015-05-28       Impact factor: 4.982

Review 2.  In Vivo NMR Studies of the Brain with Hereditary or Acquired Metabolic Disorders.

Authors:  Erica B Sherry; Phil Lee; In-Young Choi
Journal:  Neurochem Res       Date:  2015-11-26       Impact factor: 3.996

3.  Alpha-mannosidosis: correlation between phenotype, genotype and mutant MAN2B1 subcellular localisation.

Authors:  Line Borgwardt; Hilde Monica Frostad Riise Stensland; Klaus Juul Olsen; Flemming Wibrand; Helle Bagterp Klenow; Michael Beck; Yasmina Amraoui; Laila Arash; Jens Fogh; Øivind Nilssen; Christine I Dali; Allan Meldgaard Lund
Journal:  Orphanet J Rare Dis       Date:  2015-06-06       Impact factor: 4.123

Review 4.  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

5.  Use of the Bruininks-Oseretsky test of motor proficiency (BOT-2) to assess efficacy of velmanase alfa as enzyme therapy for alpha-mannosidosis.

Authors:  Dawn Phillips; Julia B Hennermann; Anna Tylki-Szymanska; Line Borgwardt; Mercedes Gil-Campos; Nathalie Guffon; Yasmina Amraoui; Silvia Geraci; Diego Ardigò; Federica Cattaneo; Allan M Lund
Journal:  Mol Genet Metab Rep       Date:  2020-04-08
  5 in total

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