Literature DB >> 31273765

Hereditary spherocytosis is associated with decreased pyruvate kinase activity due to impaired structural integrity of the red blood cell membrane.

Oliver Andres1, Felicia Loewecke1, Henner Morbach1, Sabrina Kraus2, Hermann Einsele2, Stefan Eber3, Christian P Speer1.   

Abstract

Hereditary spherocytosis (HS) is characterised by increased osmotic fragility and enhanced membrane loss of red blood cells (RBC) due to defective membrane protein complexes. In our diagnostic laboratory, we observed that pyruvate kinase (PK) activity in HS was merely slightly elevated with respect to the amount of reticulocytosis. In order to evaluate whether impaired PK activity is a feature of HS, we retrospectively analysed laboratory data sets from 172 unrelated patients with HS, hereditary elliptocytosis (HE), glucose-6-phosphate dehydrogenase (G6PD) or PK deficiency, sickle cell or haemoglobin C disease, or β-thalassaemia minor. Results from linear regression analysis provided proof that PK activity decreases with rising reticulocyte counts in HS (R2  = 0·15; slope = 9·09) and, less significantly, in HE (R2  = 0·021; slope = 8·92) when compared with other haemolytic disorders (R2  ≥ 0·65; slopes ≥ 78·6). Reticulocyte-adjusted erythrocyte PK activity levels were significantly lower in HS and even declined with increasing reticulocytes (R2  = 0·48; slope = -9·74). In this report, we describe a novel association between HS and decreased PK activity that is apparently caused by loss of membrane-bound PK due to impaired structural integrity of the RBC membrane and may aggravate severity of haemolysis in HS.
© 2019 British Society for Haematology and John Wiley & Sons Ltd.

Entities:  

Keywords:  haemolysis; pyruvate kinase deficiency; red blood cell disorders; red cell membrane; spherocytosis

Year:  2019        PMID: 31273765     DOI: 10.1111/bjh.16084

Source DB:  PubMed          Journal:  Br J Haematol        ISSN: 0007-1048            Impact factor:   6.998


  3 in total

1.  Concomitant Hereditary Spherocytosis and Pyruvate Kinase Deficiency in a Spanish Family with Chronic Hemolytic Anemia: Contribution of Laser Ektacytometry to Clinical Diagnosis.

Authors:  Joan-Lluis Vives Corrons; Elena Krishnevskaya; Laura Montllor; Valentina Leguizamon; Marta Garcia Bernal
Journal:  Cells       Date:  2022-03-28       Impact factor: 6.600

2.  Metabolic Fingerprint in Hereditary Spherocytosis Correlates With Red Blood Cell Characteristics and Clinical Severity.

Authors:  Birgit van Dooijeweert; Melissa H Broeks; Nanda M Verhoeven-Duif; Wouter W van Solinge; Eduard J van Beers; Minke A E Rab; Edward E S Nieuwenhuis; Judith J M Jans; Marije Bartels; Richard van Wijk
Journal:  Hemasphere       Date:  2021-06-12

Review 3.  Molecular heterogeneity of pyruvate kinase deficiency.

Authors:  Paola Bianchi; Elisa Fermo
Journal:  Haematologica       Date:  2020-09-01       Impact factor: 9.941

  3 in total

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