Literature DB >> 23233556

The porphyrias: advances in diagnosis and treatment.

Manisha Balwani1, Robert J Desnick.   

Abstract

The inborn errors of heme biosynthesis, the porphyrias, are 8 genetically distinct metabolic disorders that can be classified as "acute hepatic," "hepatic cutaneous," and "erythropoietic cutaneous" diseases. Recent advances in understanding their pathogenesis and molecular genetic heterogeneity have led to improved diagnosis and treatment. These advances include DNA-based diagnoses for all the porphyrias, new understanding of the pathogenesis of the acute hepatic porphyrias, identification of the iron overload-induced inhibitor of hepatic uroporphyrin decarboxylase activity that causes the most common porphyria, porphyria cutanea tarda, the identification of an X-linked form of erythropoietic protoporphyria due to gain-of-function mutations in erythroid-specific 5-aminolevulinate synthase (ALAS2), and new and experimental treatments for the erythropoietic prophyrias. Knowledge of these advances is relevant for hematologists because they administer the hematin infusions to treat the acute attacks in patients with the acute hepatic porphyrias, perform the chronic phlebotomies to reduce the iron overload and clear the dermatologic lesions in porphyria cutanea tarda, and diagnose and treat the erythropoietic porphyrias, including chronic erythrocyte transfusions, bone marrow or hematopoietic stem cell transplants, and experimental pharmacologic chaperone and stem cell gene therapies for congenital erythropoietic protoporphyria. These developments are reviewed to update hematologists on the latest advances in these diverse disorders.

Entities:  

Mesh:

Substances:

Year:  2012        PMID: 23233556     DOI: 10.1182/asheducation-2012.1.19

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


  7 in total

Review 1.  Modelling inborn errors of metabolism in zebrafish.

Authors:  Kim Wager; Fahad Mahmood; Claire Russell
Journal:  J Inherit Metab Dis       Date:  2014-05-06       Impact factor: 4.982

2.  Acute intermittent porphyria: prevalence of pathogenic HMBS variants in China, and epidemiological survey in Hebei Province, China.

Authors:  Liyan Ma; Yu Tian; Xuan Qi; Pei Li; Jie Li; Qing Teng; Yuelin Ma; Songyun Zhang
Journal:  Ann Transl Med       Date:  2022-05

3.  N-alkylprotoporphyrin formation and hepatic porphyria in dogs after administration of a new antiepileptic drug candidate: mechanism and species specificity.

Authors:  Jean-Marie Nicolas; Hugues Chanteux; Valérie Mancel; Guy-Marie Dubin; Brigitte Gerin; Ludovicus Staelens; Olympe Depelchin; Sophie Kervyn
Journal:  Toxicol Sci       Date:  2014-06-27       Impact factor: 4.849

4.  The essential role of the transporter ABCG2 in the pathophysiology of erythropoietic protoporphyria.

Authors:  Pengcheng Wang; Madhav Sachar; Jie Lu; Amina I Shehu; Junjie Zhu; Jing Chen; Ke Liu; Karl E Anderson; Wen Xie; Frank J Gonzalez; Curtis D Klaassen; Xiaochao Ma
Journal:  Sci Adv       Date:  2019-09-18       Impact factor: 14.136

5.  Delivery of oligonucleotides to bone marrow to modulate ferrochelatase splicing in a mouse model of erythropoietic protoporphyria.

Authors:  François Halloy; Pavithra S Iyer; Paulina Ćwiek; Alice Ghidini; Jasmin Barman-Aksözen; Nicole Wildner-Verhey van Wijk; Alexandre P A Theocharides; Elisabeth I Minder; Xiaoye Schneider-Yin; Daniel Schümperli; Jonathan Hall
Journal:  Nucleic Acids Res       Date:  2020-05-21       Impact factor: 16.971

Review 6.  Biology of Heme in Mammalian Erythroid Cells and Related Disorders.

Authors:  Tohru Fujiwara; Hideo Harigae
Journal:  Biomed Res Int       Date:  2015-10-18       Impact factor: 3.411

7.  Fundamentals of G-quadruplex biology.

Authors:  F Brad Johnson
Journal:  Annu Rep Med Chem       Date:  2020-07-30       Impact factor: 1.059

  7 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.