Literature DB >> 24739649

Congenital disorders of glycosylation with neonatal presentation.

Catarina Resende1, Carmen Carvalho, Artur Alegria, Dulce Oliveira, Dulce Quelhas, Anabela Bandeira, Elisa Proença.   

Abstract

Congenital disorders of glycosylation (CDG) are a group of hereditary diseases characterised by deficiency of enzymes involved in proteins glycosylation. We describe the clinical case of a neonate with CDG type 1a, nowadays designated phosphomannomutase 2 (PMM2)-CDG. Physical examination showed an abnormal facies, axial hypotonia, abnormal fat distribution, inverted nipples, non-palpable testicles and arachnodactyly. Progressive multiple system organ involvement and worsening of hypertrophic cardiomyopathy occurred. Metabolic study revealed a CDG disturbance, which was confirmed by genetic study. The following mutations were identified: c.193G>T; p.D65Y and c.470T>C; p.F157S. Clinical deterioration was inevitable with multisystemic failure and death. CDG represents a challenge for physicians due to multiple organ involvement, and heterogeneous clinical manifestations. The neonatal form is usually associated with the worst prognosis.

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Year:  2014        PMID: 24739649      PMCID: PMC3992553          DOI: 10.1136/bcr-2013-010037

Source DB:  PubMed          Journal:  BMJ Case Rep        ISSN: 1757-790X


  5 in total

1.  Phosphomannose isomerase inhibitors improve N-glycosylation in selected phosphomannomutase-deficient fibroblasts.

Authors:  Vandana Sharma; Mie Ichikawa; Ping He; David A Scott; Yalda Bravo; Russell Dahl; Bobby G Ng; Nicholas D P Cosford; Hudson H Freeze
Journal:  J Biol Chem       Date:  2011-09-26       Impact factor: 5.157

2.  Congenital disorder of glycosylation type Ia: searching for the origin of common mutations in PMM2.

Authors:  D Quelhas; R Quental; L Vilarinho; A Amorim; L Azevedo
Journal:  Ann Hum Genet       Date:  2006-12-12       Impact factor: 1.670

3.  Unusual presentation of congenital disorder of glycosylation type 1a: congenital persistent thrombocytopenia, hypertrophic cardiomyopathy and hydrops-like aspect due to marked peripheral oedema.

Authors:  Vera Noelle; Matthias Knuepfer; Ferdinand Pulzer; Volker Schuster; Werner Siekmeyer; Gert Matthijs; Christoph Vogtmann
Journal:  Eur J Pediatr       Date:  2005-01-12       Impact factor: 3.183

Review 4.  The clinical spectrum of phosphomannomutase 2 deficiency (CDG-Ia).

Authors:  Stephanie Grünewald
Journal:  Biochim Biophys Acta       Date:  2009-01-14

5.  Hypertrophic obstructive cardiomyopathy in a neonate with the carbohydrate-deficient glycoprotein syndrome.

Authors:  P T Clayton; B G Winchester; G Keir
Journal:  J Inherit Metab Dis       Date:  1992       Impact factor: 4.982

  5 in total
  4 in total

Review 1.  Recognizable phenotypes in CDG.

Authors:  Carlos R Ferreira; Ruqaia Altassan; Dorinda Marques-Da-Silva; Rita Francisco; Jaak Jaeken; Eva Morava
Journal:  J Inherit Metab Dis       Date:  2018-04-13       Impact factor: 4.982

Review 2.  Cardiac complications of congenital disorders of glycosylation (CDG): a systematic review of the literature.

Authors:  D Marques-da-Silva; R Francisco; D Webster; V Dos Reis Ferreira; J Jaeken; T Pulinilkunnil
Journal:  J Inherit Metab Dis       Date:  2017-07-19       Impact factor: 4.982

3.  Cystic kidney diseases associated with mutations in phosphomannomutase 2 promotor: a large spectrum of phenotypes.

Authors:  Guillaume Dorval; Cécile Jeanpierre; Vincent Morinière; Carole Tournant; Bettina Bessières; Tania Attié-Bittach; Jeanne Amiel; Emmanuel Spaggari; Yves Ville; Elodie Merieau; Marie-Claire Gubler; Sophie Saunier; Laurence Heidet
Journal:  Pediatr Nephrol       Date:  2021-02-13       Impact factor: 3.714

4.  Clinical and radiological correlates of activities of daily living in cerebellar atrophy caused by PMM2 mutations (PMM2-CDG).

Authors:  Fabio Pettinato; Giovanni Mostile; Roberta Battini; Diego Martinelli; Annalisa Madeo; Elisa Biamino; Daniele Frattini; Domenico Garozzo; Serena Gasperini; Rossella Parini; Fabio Sirchia; Giuseppe Sortino; Luisa Sturiale; Gert Matthijs; Amelia Morrone; Maja Di Rocco; Renata Rizzo; Jaak Jaeken; Agata Fiumara; Rita Barone
Journal:  Cerebellum       Date:  2021-02-22       Impact factor: 3.847

  4 in total

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