Your browser doesn't support javascript.
Mostrar: 20 | 50 | 100
Resultados 1 - 1 de 1
Filtrar
Adicionar filtros

Base de dados
Ano de publicação
Tipo de documento
Intervalo de ano
1.
Mol Genet Metab ; 138(4): 107559, 2023 04.
Artigo em Inglês | MEDLINE | ID: covidwho-20243551

RESUMO

Phosphomannomutase-2-congenital disorder of glycosylation (PMM2-CDG) is the most common CDG and presents with highly variable features ranging from isolated neurologic involvement to severe multi-organ dysfunction. Liver abnormalities occur in in almost all patients and frequently include hepatomegaly and elevated aminotransferases, although only a minority of patients develop progressive hepatic fibrosis and liver failure. No curative therapies are currently available for PMM2-CDG, although investigation into several novel therapies is ongoing. We report the first successful liver transplantation in a 4-year-old patient with PMM2-CDG. Over a 3-year follow-up period, she demonstrated improved growth and neurocognitive development and complete normalization of liver enzymes, coagulation parameters, and carbohydrate-deficient transferrin profile, but persistently abnormal IgG glycosylation and recurrent upper airway infections that did not require hospitalization. Liver transplant should be considered as a treatment option for PMM2-CDG patients with end-stage liver disease, however these patients may be at increased risk for recurrent bacterial infections post-transplant.


Assuntos
Defeitos Congênitos da Glicosilação , Transplante de Fígado , Fosfotransferases (Fosfomutases) , Feminino , Humanos , Pré-Escolar , Glicosilação , Seguimentos , Fosfotransferases (Fosfomutases)/genética , Defeitos Congênitos da Glicosilação/complicações , Defeitos Congênitos da Glicosilação/genética , Fígado/metabolismo , Imunoglobulina G
SELEÇÃO DE REFERÊNCIAS
DETALHE DA PESQUISA