Literature DB >> 33275677

Updates on the diagnosis and management of the most common hereditary porphyrias: AIP and EPP.

Michael Linenberger1, Kleber Y Fertrin1.   

Abstract

The porphyrias are a family of metabolic disorders caused by defects in the activity of one of the enzymes in the heme biosynthetic pathway. Acute intermittent porphyria (AIP), caused by autosomal dominant mutations in the gene encoding hydroxymethylbilane synthase, can lead to hepatocyte overaccumulation and systemic distribution of the proximal porphyrin precursors, 5-aminolevulinic acid (ALA) and porphobilinogen (PBG). ALA and PBG are toxic to neurons and extrahepatic tissue and cause the neurovisceral clinical manifestations of AIP. Management of AIP includes awareness and avoidance of triggering factors, infusions of hemin for severe acute attacks, and, if indicated for chronic suppressive therapy, maintenance treatment with hemin or givosiran, a small interfering RNA molecule that antagonizes ALA synthase 1 transcripts. Erythropoietic protoporphyria (EPP) is most commonly caused by autosomal recessive mutations in the gene encoding ferrochelatase (FECH), the heme pathway terminal enzyme. FECH deficiency leads to erythrocyte overaccumulation and high plasma levels of lipophilic protoporphyrins that photoactivate in the skin, causing burning pain and erythema. Protoporphyrins excreted in the bile can cause gallstones, cholestasis, fibrosis, and ultimately liver failure. Management of EPP includes skin protection and afamelanotide, an α-melanocyte stimulating hormone analog that increases melanin pigment and reduces photoactivation. Liver transplantation may be necessary for severe EPP-induced liver complications. Because AIP and EPP arise from defects in the heme biosynthetic pathway, hematologists are often consulted to evaluate and manage suspected or proven porphyrias. A working knowledge of these disorders increases our confidence and effectiveness as consultants and medical providers.
© 2020 by The American Society of Hematology.

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Year:  2020        PMID: 33275677      PMCID: PMC7727547          DOI: 10.1182/hematology.2020000124

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


  33 in total

1.  Clinical, Biochemical, and Genetic Characterization of North American Patients With Erythropoietic Protoporphyria and X-linked Protoporphyria.

Authors:  Manisha Balwani; Hetanshi Naik; Karl E Anderson; D Montgomery Bissell; Joseph Bloomer; Herbert L Bonkovsky; John D Phillips; Jessica R Overbey; Bruce Wang; Ashwani K Singal; Lawrence U Liu; Robert J Desnick
Journal:  JAMA Dermatol       Date:  2017-08-01       Impact factor: 10.282

2.  The role for BMT in erythropoietic protoporphyria.

Authors:  S Wahlin; P Harper
Journal:  Bone Marrow Transplant       Date:  2009-06-15       Impact factor: 5.483

Review 3.  Porphyria.

Authors:  D Montgomery Bissell; Karl E Anderson; Herbert L Bonkovsky
Journal:  N Engl J Med       Date:  2017-08-31       Impact factor: 91.245

4.  Plasma and red cell exchange transfusions for erythropoietic protoporphyria: a case report and review of the literature.

Authors:  Monica B Pagano; William Hobbs; Michael Linenberger; Meghan Delaney
Journal:  J Clin Apher       Date:  2012-09-21       Impact factor: 2.821

5.  Results of a pilot study of isoniazid in patients with erythropoietic protoporphyria.

Authors:  Charles J Parker; Robert J Desnick; Montgomery D Bissel; Joseph R Bloomer; Ashwani Singal; Laurent Gouya; Herve Puy; Karl E Anderson; Manisha Balwani; John D Phillips
Journal:  Mol Genet Metab       Date:  2019-07-31       Impact factor: 4.797

Review 6.  Pathogenesis and clinical features of the acute hepatic porphyrias (AHPs).

Authors:  Herbert L Bonkovsky; Natalia Dixon; Sean Rudnick
Journal:  Mol Genet Metab       Date:  2019-03-06       Impact factor: 4.797

Review 7.  Acute hepatic porphyrias: Current diagnosis & management.

Authors:  Karl E Anderson
Journal:  Mol Genet Metab       Date:  2019-07-05       Impact factor: 4.797

8.  International Porphyria Molecular Diagnostic Collaborative: an evidence-based database of verified pathogenic and benign variants for the porphyrias.

Authors:  Brenden Chen; Sharon Whatley; Michael Badminton; Aasne K Aarsand; Karl E Anderson; D Montgomery Bissell; Herbert L Bonkovsky; Maria D Cappellini; Ylva Floderus; Edith C H Friesema; Laurent Gouya; Pauline Harper; Raili Kauppinen; Yonina Loskove; Pavel Martásek; John D Phillips; Hervé Puy; Sverre Sandberg; Caroline Schmitt; Jordi To-Figueras; Yedidyah Weiss; Makiko Yasuda; Jean-Charles Deybach; Robert J Desnick
Journal:  Genet Med       Date:  2019-05-10       Impact factor: 8.822

9.  Medical and financial burden of acute intermittent porphyria.

Authors:  Rochus A Neeleman; Margreet A E M Wagenmakers; Rita H Koole-Lesuis; G Sophie Mijnhout; J H Paul Wilson; Edith C H Friesema; Janneke G Langendonk
Journal:  J Inherit Metab Dis       Date:  2018-04-19       Impact factor: 4.982

10.  EXPLORE: A Prospective, Multinational, Natural History Study of Patients with Acute Hepatic Porphyria with Recurrent Attacks.

Authors:  Laurent Gouya; Paolo Ventura; Manisha Balwani; D Montgomery Bissell; David C Rees; Ulrich Stölzel; John D Phillips; Raili Kauppinen; Janneke G Langendonk; Robert J Desnick; Jean-Charles Deybach; Herbert L Bonkovsky; Charles Parker; Hetanshi Naik; Michael Badminton; Penelope E Stein; Elisabeth Minder; Jerzy Windyga; Radan Bruha; Maria Domenica Cappellini; Eliane Sardh; Pauline Harper; Sverre Sandberg; Aasne K Aarsand; Janice Andersen; Félix Alegre; Aneta Ivanova; Neila Talbi; Amy Chan; William Querbes; John Ko; Craig Penz; Shangbin Liu; Tim Lin; Amy Simon; Karl E Anderson
Journal:  Hepatology       Date:  2019-11-07       Impact factor: 17.425

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  3 in total

Review 1.  Dosage Compensation in Females with X-Linked Metabolic Disorders.

Authors:  Patrycja Juchniewicz; Ewa Piotrowska; Anna Kloska; Magdalena Podlacha; Jagoda Mantej; Grzegorz Węgrzyn; Stefan Tukaj; Joanna Jakóbkiewicz-Banecka
Journal:  Int J Mol Sci       Date:  2021-04-26       Impact factor: 5.923

2.  Erythropoietic Protoporphyria: You May Not Have Seen It, but It May Have Seen You.

Authors:  Larissa M Pastore; Christina W Sun; Sylvia Hsu
Journal:  Cureus       Date:  2022-03-17

Review 3.  Cutting-Edge Therapies and Novel Strategies for Acute Intermittent Porphyria: Step-by-Step towards the Solution.

Authors:  Miriam Longo; Erika Paolini; Marica Meroni; Paola Dongiovanni
Journal:  Biomedicines       Date:  2022-03-11
  3 in total

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