Literature DB >> 30681718

How we manage patients with pyruvate kinase deficiency.

Rachael F Grace1, D Mark Layton2, Wilma Barcellini3.   

Abstract

Novel therapies in development have brought a new focus on pyruvate kinase deficiency (PKD), the most common congenital haemolytic anaemia due to a glycolytic enzyme deficiency. With an improved recognition of its clinical presentation and understanding of the diagnostic pathway, more patients are likely to be identified with this anaemia. Complications, including gallstones and non-transfusion-related iron overload, require monitoring for early diagnosis and management. Current management remains supportive with red cell transfusions, chelation and splenectomy. Decisions to transfuse and/or splenectomise must be individualised. Haematopoietic stem cell transplant has been pursued in a small number of patients with mixed outcomes. Novel treatment approaches, which range from a small molecule pyruvate kinase activator to gene therapy, may transform the way in which PKD is managed in the future. In this review, we discuss the pathophysiology of PKD and present our approaches to diagnosis, monitoring and management of patients with this anaemia.
© 2019 British Society for Haematology and John Wiley & Sons Ltd.

Entities:  

Keywords:  Pyruvate kinase deficiency; congenital haemolytic anaemia; iron overload; splenectomy; transfusions

Year:  2019        PMID: 30681718     DOI: 10.1111/bjh.15758

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


  14 in total

1.  Erythrocytapheresis as a novel treatment option for adult patients with pyruvate kinase deficiency.

Authors:  Rawia F G Jensen; Morten H Dziegiel; Klaus Rieneck; Henrik Birgens; Andreas Glenthøj
Journal:  Haematologica       Date:  2020-04-09       Impact factor: 9.941

2.  Genotype-phenotype correlation and molecular heterogeneity in pyruvate kinase deficiency.

Authors:  Paola Bianchi; Elisa Fermo; Kimberly Lezon-Geyda; Eduard J van Beers; Holmes D Morton; Wilma Barcellini; Bertil Glader; Satheesh Chonat; Yaddanapudi Ravindranath; Peter E Newburger; Nina Kollmar; Jenny M Despotovic; Madeleine Verhovsek; Mukta Sharma; Janet L Kwiatkowski; Kevin H M Kuo; Marcin W Wlodarski; Hassan M Yaish; Susanne Holzhauer; Heng Wang; Joachim Kunz; Kathryn Addonizio; Hasan Al-Sayegh; Wendy B London; Oliver Andres; Richard van Wijk; Patrick G Gallagher; Rachael F F Grace
Journal:  Am J Hematol       Date:  2020-03-06       Impact factor: 10.047

Review 3.  The Interplay between Drivers of Erythropoiesis and Iron Homeostasis in Rare Hereditary Anemias: Tipping the Balance.

Authors:  Simon Grootendorst; Jonathan de Wilde; Birgit van Dooijeweert; Annelies van Vuren; Wouter van Solinge; Roger Schutgens; Richard van Wijk; Marije Bartels
Journal:  Int J Mol Sci       Date:  2021-02-23       Impact factor: 5.923

Review 4.  Genetics and Genomics Approaches for Diagnosis and Research Into Hereditary Anemias.

Authors:  Roberta Russo; Roberta Marra; Barbara Eleni Rosato; Achille Iolascon; Immacolata Andolfo
Journal:  Front Physiol       Date:  2020-12-22       Impact factor: 4.566

5.  Comorbidities and complications in adults with pyruvate kinase deficiency.

Authors:  Audra N Boscoe; Yan Yan; Elizabeth Hedgeman; Eduard J van Beers; Hanny Al-Samkari; Wilma Barcellini; Stefan W Eber; Bertil Glader; Hassan M Yaish; Satheesh Chonat; Mukta Sharma; Kevin H M Kuo; Ellis J Neufeld; Heng Wang; Madeleine Verhovsek; Sujit Sheth; Rachael F Grace
Journal:  Eur J Haematol       Date:  2021-01-24       Impact factor: 2.997

6.  A Proposed Concept for Defective Mitophagy Leading to Late Stage Ineffective Erythropoiesis in Pyruvate Kinase Deficiency.

Authors:  Annelies Johanna van Vuren; Eduard Johannes van Beers; Richard van Wijk
Journal:  Front Physiol       Date:  2021-01-20       Impact factor: 4.566

Review 7.  Mitapivat, a novel pyruvate kinase activator, for the treatment of hereditary hemolytic anemias.

Authors:  Hanny Al-Samkari; Eduard J van Beers
Journal:  Ther Adv Hematol       Date:  2021-12-21

8.  A novel homozygous missense variant p.D339N in the PKLR gene correlates with pyruvate kinase deficiency in a Pakistani family: a case report.

Authors:  Atta Ur Rehman; Abdur Rashid; Zubair Hussain; Khadim Shah
Journal:  J Med Case Rep       Date:  2022-02-16

9.  Splenectomy and emerging novel treatments in rare inherited hemolytic anemias.

Authors:  Joanne Yacobovich; Hannah Tamary
Journal:  Hemasphere       Date:  2019-06-30

10.  The variable manifestations of disease in pyruvate kinase deficiency and their management.

Authors:  Hanny Al-Samkari; Eduard J Van Beers; Kevin H M Kuo; Wilma Barcellini; Paola Bianchi; Andreas Glenthøj; María Del Mar Mañú Pereira; Richard Van Wijk; Bertil Glader; Rachael F Grace
Journal:  Haematologica       Date:  2020-09-01       Impact factor: 9.941

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