| Literature DB >> 24739649 |
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.Entities:
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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