Pyruvate kinase deficiency
Pyruvate kinase deficiency is the most common inherited glycolytic enzyme defect causing chronic hemolytic anemia, an autosomal recessive disorder in which ATP-depleted, rigid erythrocytes are cleared extravascularly by the spleen. Diagnosis rests on a reduced red cell pyruvate kinase enzyme activity assay or genetic testing in a patient with lifelong nonspherocytic hemolysis, echinocytes on smear, and a negative direct antiglobulin test, and management ranges from folic acid alone to transfusion, splenectomy, or the pyruvate kinase activator mitapivat.
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What this article covers
- Overview
Definition: pyruvate kinase deficiency is an autosomal recessive defect of the terminal glycolytic enzyme pyruvate kinase in erythrocytes, producing chronic, lifelong hemolytic anemia.
- Epidemiology
Rare but the leading glycolytic cause: an uncommon disease overall, but the most frequently identified inherited glycolytic pathway defect causing hemolytic anemia, more common in persons of Northern and Eastern European descent.
- Risk factors
Biallelic PKLR mutations: homozygous or compound heterozygous pathogenic variants in the gene encoding red cell pyruvate kinase are required for clinically significant disease; heterozygous carriers are asymptomatic.
- Pathophysiology
Glycolytic block at the final step: pyruvate kinase catalyzes the last step of glycolysis, converting phosphoenolpyruvate to pyruvate while generating ATP; because mature erythrocytes lack mitochondria, glycolysis is their sole ATP source.
- Clinical features
Chronic hemolytic anemia from birth or early childhood: the hallmark presentation, with jaundice frequently apparent in the neonatal period and sometimes severe enough to require exchange transfusion for hyperbilirubinemia.
- Differential diagnosis
Glucose-6-phosphate dehydrogenase deficiency: X-linked, episodic oxidant-triggered hemolysis after drugs, infection, or fava beans; smear shows bite cells and Heinz bodies rather than echinocytes…
- Diagnosis
Establish the diagnosis with a reduced red cell pyruvate kinase enzyme activity assay and/or PKLR genetic testing in a patient with chronic nonspherocytic hemolytic anemia and normal G6PD activity…
- Management
Start folic acid in every patient with chronic hemolysis: folic acid 1 to 5 mg orally once daily, continued indefinitely while hemolysis is ongoing, to support the compensatory erythropoiesis driven by chronic red cell turnover.
- Complications
Iron overload and end-organ damage: chronic hemolysis increases intestinal iron absorption independent of transfusion, and repeated transfusion accelerates it, causing cardiac, hepatic, and endocrine damage if unmonitored.
- Prognosis
Highly variable, genotype-dependent course: ranges from severe transfusion-dependent disease presenting in the neonatal period to mild, fully compensated hemolysis discovered incidentally in an asymptomatic adult.
- Prevention
Genetic counseling for known carriers or affected families: offer counseling and PKLR carrier testing to parents of an affected child and at-risk relatives; recurrence risk in a future pregnancy of two carrier parents is 25%.
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Related articles
- Glucose-6-phosphate dehydrogenase deficiency
Glucose-6-phosphate dehydrogenase (G6PD) deficiency is an X-linked enzymopathy that leaves red cells unable to buffer oxidative stress, producing episodic intravascular hemolysis with bite cells and Heinz bodies after infection, fava beans, or oxidant drugs. Diagnosis rests on a G6PD enzyme activity assay that can be falsely normal during the acute episode and must be repeated 2 to 3 months later, while management centers on trigger avoidance, supportive care, and transfusion for severe hemolysis.
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- Alpha-thalassemia
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- Paroxysmal nocturnal hemoglobinuria
Paroxysmal nocturnal hemoglobinuria is an acquired clonal stem cell disorder caused by a somatic PIGA mutation that leaves blood cells unprotected from complement, producing intravascular hemolysis, unusual-site venous thrombosis, and cytopenias. Diagnosis rests on flow cytometry showing loss of the GPI-anchored proteins CD55 and CD59, and eculizumab or ravulizumab is used to block terminal complement and reduce both hemolysis and thrombotic risk, while allogeneic stem cell transplant remains the only cure.