Literature DB >> 34889365

Diagnosis and clinical management of enzymopathies.

Lucio Luzzatto1.   

Abstract

At least 16 genetically determined conditions qualify as red blood cell enzymopathies. They range in frequency from ultrarare to rare, with the exception of glucose-6-phosphate dehydrogenase deficiency, which is very common. Nearly all these enzymopathies manifest as chronic hemolytic anemias, with an onset often in the neonatal period. The diagnosis can be quite easy, such as when a child presents with dark urine after eating fava beans, or it can be quite difficult, such as when an adult presents with mild anemia and gallstones. In general, 4 steps are recommended: (1) recognizing chronic hemolytic anemia; (2) excluding acquired causes; (3) excluding hemoglobinopathies and membranopathies; (4) pinpointing which red blood cell enzyme is deficient. Step 4 requires 1 or many enzyme assays; alternatively, DNA testing against an appropriate gene panel can combine steps 3 and 4. Most patients with a red blood cell enzymopathy can be managed by good supportive care, including blood transfusion, iron chelation when necessary, and splenectomy in selected cases; however, some patients have serious extraerythrocytic manifestations that are difficult to manage. In the absence of these, red blood cell enzymopathies are in principle amenable to hematopoietic stem cell transplantation and gene therapy/gene editing.
Copyright © 2021 by The American Society of Hematology.

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Year:  2021        PMID: 34889365      PMCID: PMC8791163          DOI: 10.1182/hematology.2021000266

Source DB:  PubMed          Journal:  Hematology Am Soc Hematol Educ Program        ISSN: 1520-4383


  49 in total

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2.  A Rare Cause of Neonatal Hemolytic Anemia: Glutathione Synthetase Deficiency.

Authors:  Pembe Soylu Ustkoyuncu; Fatma Türkan Mutlu; Aslihan Kiraz; Zuhal Tag Balkis; Sibel Yel
Journal:  J Pediatr Hematol Oncol       Date:  2018-01       Impact factor: 1.289

3.  Erythrocyte glucose 6-phosphate dehydrogenase deficiency (G6PD type A-) and neonatal jaundice.

Authors:  U Bienzle; C Effiong; L Luzzatto
Journal:  Acta Paediatr Scand       Date:  1976-11

4.  Glucose-6-phosphate dehydrogenase deficiency.

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Journal:  Blood       Date:  2020-09-10       Impact factor: 22.113

5.  Management of pyruvate kinase deficiency in children and adults.

Authors:  Rachael F Grace; Wilma Barcellini
Journal:  Blood       Date:  2020-09-10       Impact factor: 22.113

6.  Mortality in a cohort of men expressing the glucose-6-phosphate dehydrogenase deficiency.

Authors:  P Cocco; P Todde; S Fornera; M B Manca; P Manca; A R Sias
Journal:  Blood       Date:  1998-01-15       Impact factor: 22.113

7.  The need for neonatal glucose-6-phosphate dehydrogenase screening: a global perspective.

Authors:  M Kaplan; C Hammerman
Journal:  J Perinatol       Date:  2009-02       Impact factor: 2.521

8.  Molecular cloning and expression of human liver biliverdin-IX beta reductase.

Authors:  A Komuro; T Tobe; K Hashimoto; Y Nakano; T Yamaguchi; H Nakajima; M Tomita
Journal:  Biol Pharm Bull       Date:  1996-06       Impact factor: 2.233

9.  Molecular characterization and mapping of glucose-6-phosphate dehydrogenase (G6PD) mutations in the Greater Mekong Subregion.

Authors:  Germana Bancone; Didier Menard; Nimol Khim; Saorin Kim; Lydie Canier; Chea Nguong; Koukeo Phommasone; Mayfong Mayxay; Sabine Dittrich; Malavanh Vongsouvath; Nadine Fievet; Jean-Yves Le Hesran; Valerie Briand; Sommay Keomany; Paul N Newton; Gornpan Gorsawun; Kaelan Tardy; Cindy S Chu; Orpreeya Rattanapalroj; Le Thanh Dong; Huynh Hong Quang; Nguyen Tam-Uyen; Nguyen Thuy-Nhien; Tran Tinh Hien; Michael Kalnoky; Francois Nosten
Journal:  Malar J       Date:  2019-01-23       Impact factor: 2.979

10.  Evaluation of a Novel Quantitative Test for Glucose-6-Phosphate Dehydrogenase Deficiency: Bringing Quantitative Testing for Glucose-6-Phosphate Dehydrogenase Deficiency Closer to the Patient.

Authors:  Sampa Pal; Pooja Bansil; Germana Bancone; Sevan Hrutkay; Maria Kahn; Gornpan Gornsawun; Pimsupah Penpitchaporn; Cindy S Chu; François Nosten; Gonzalo J Domingo
Journal:  Am J Trop Med Hyg       Date:  2019-01       Impact factor: 2.345

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