Literature DB >> 17451957

Congenital disorder of glycosylation type 1a: three siblings with a mild neurological phenotype.

D Coman1, J McGill, R MacDonald, D Morris, S Klingberg, J Jaeken, D Appleton.   

Abstract

We report 3 siblings (1 male and 2 female) recently diagnosed with congenital disorder of glycosylation type Ia (CDG-Ia) in their mid-20s. They experience mild mental retardation but manage to function independently in society. Their professions are library assistant, professional artistic painter and secretarial work. All three siblings have cerebellar hypoplasia and ataxia, but are able to ambulate easily. Two of the siblings have required strabismus surgical repairs. All have antithrombin III deficiency, osteoporosis, and mild dysmorphic features. Hypergonadotrophic hypogonadism was a feature of the two female siblings. A type 1 sialotransferrin pattern and phosphomannomutase (PMM) deficiency have been demonstrated. They are compound heterozygotes for R141H and L32R mutations in the PMM2 gene. While there is clinical heterogeneity in CDG-Ia, we believe that our patients are among the mildest of intellectually affected CDG-Ia patients reported to date.

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Year:  2007        PMID: 17451957     DOI: 10.1016/j.jocn.2006.04.008

Source DB:  PubMed          Journal:  J Clin Neurosci        ISSN: 0967-5868            Impact factor:   1.961


  14 in total

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Review 2.  Congenital disorders of glycosylation.

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Authors:  Rita Barone; M Carrozzi; R Parini; R Battini; D Martinelli; M Elia; M Spada; F Lilliu; G Ciana; A Burlina; V Leuzzi; M Leoni; L Sturiale; G Matthijs; J Jaeken; M Di Rocco; D Garozzo; A Fiumara
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7.  A mouse model of a human congenital disorder of glycosylation caused by loss of PMM2.

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8.  Congenital protein hypoglycosylation diseases.

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Review 9.  Insights into complexity of congenital disorders of glycosylation.

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Journal:  Biochem Med (Zagreb)       Date:  2012       Impact factor: 2.313

10.  29 French adult patients with PMM2-congenital disorder of glycosylation: outcome of the classical pediatric phenotype and depiction of a late-onset phenotype.

Authors:  Marie-Lorraine Monin; Cyril Mignot; Pascale De Lonlay; Bénédicte Héron; Alice Masurel; Michèle Mathieu-Dramard; Catherine Lenaerts; Christel Thauvin; Marion Gérard; Emmanuel Roze; Aurélia Jacquette; Perrine Charles; Claire de Baracé; Valérie Drouin-Garraud; Philippe Khau Van Kien; Valérie Cormier-Daire; Michèle Mayer; Hélène Ogier; Alexis Brice; Nathalie Seta; Delphine Héron
Journal:  Orphanet J Rare Dis       Date:  2014-12-11       Impact factor: 4.123

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