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Mol Genet Metab ; 138(4): 107559, 2023 04.
Article in English | MEDLINE | ID: covidwho-20243551

ABSTRACT

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.


Subject(s)
Congenital Disorders of Glycosylation , Liver Transplantation , Phosphotransferases (Phosphomutases) , Female , Humans , Child, Preschool , Glycosylation , Follow-Up Studies , Phosphotransferases (Phosphomutases)/genetics , Congenital Disorders of Glycosylation/complications , Congenital Disorders of Glycosylation/genetics , Liver/metabolism , Immunoglobulin G
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