Journal of human genetics · 2020
Novel missense variants in PCK1 gene cause cytosolic PEPCK deficiency with growth failure from inadequate caloric intake.
Клинический случайСтатьяНазвание и аннотация: Перевод в очереди
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Oishi Kimihiko, Siegel Casey, Cork Emalyn E, Chen Hongjie, Imagawa Eri
Аннотация
Cytosolic PEPCK deficiency (PCKDC) is a rare autosomal recessive inborn error of metabolism, which can present with hypoglycemia, lactic acidosis, and liver failure. It is caused by biallelic pathogenic variants in the PCK1 gene. Only four PCK1 variants have been previously reported in seven patients with PCKDC, and their clinical course of this condition has not been well characterized. Here, we report a Hispanic male with novel biallelic PCK1 variants, p.(Gly430Asp) and p.(His496Gln), who had a unique clinical presentation. He presented with a new onset of growth failure, elevated blood lactate, transaminitis, and abnormal urine metabolites profile, but he has not had documented hypoglycemia. Growth restriction happened due to insufficient caloric intake, and it was improved with nutritional intervention. PCKDC is a manageable disorder and therefore appropriate nutritional and clinical suspicion from typical lab abnormalities which lead to molecular confirmation tests are essential to prevent poor clinical outcomes.
Предметные рубрики MeSH
Amino Acid SequenceBirth WeightChild, PreschoolCitric Acid CycleCodon, NonsenseCytosolEnergy IntakeFailure to ThriveFemaleFood PreferencesGenotypeGrowth DisordersHumansInfant FoodIntracellular Signaling Peptides and ProteinsMaleMicrocephalyPedigreePhosphoenolpyruvate Carboxykinase (GTP)PregnancyPregnancy ComplicationsSeizuresSequence AlignmentSequence Homology, Amino Acid