Literature DB >> 31073229

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

Brenden Chen1, Sharon Whatley2, Michael Badminton3, Aasne K Aarsand4, Karl E Anderson5, D Montgomery Bissell6, Herbert L Bonkovsky7, Maria D Cappellini8, Ylva Floderus9, Edith C H Friesema10, Laurent Gouya11, Pauline Harper9, Raili Kauppinen12, Yonina Loskove1, Pavel Martásek13, John D Phillips14, Hervé Puy11, Sverre Sandberg4,15, Caroline Schmitt11, Jordi To-Figueras16, Yedidyah Weiss1, Makiko Yasuda1, Jean-Charles Deybach17, Robert J Desnick18.   

Abstract

With the advent of precision and genomic medicine, a critical issue is whether a disease gene variant is pathogenic or benign. Such is the case for the three autosomal dominant acute hepatic porphyrias (AHPs), including acute intermittent porphyria, hereditary coproporphyria, and variegate porphyria, each resulting from the half-normal enzymatic activities of hydroxymethylbilane synthase, coproporphyrinogen oxidase, and protoporphyrinogen oxidase, respectively. To date, there is no public database that documents the likely pathogenicity of variants causing the porphyrias, and more specifically, the AHPs with biochemically and clinically verified information. Therefore, an international collaborative with the European Porphyria Network and the National Institutes of Health/National Center for Advancing Translational Sciences/National Institute of Diabetes and Digestive and Kidney Diseases (NIH/NCATS/NIDDK)-sponsored Porphyrias Consortium of porphyria diagnostic experts is establishing an online database that will collate biochemical and clinical evidence verifying the pathogenicity of the published and newly identified variants in the AHP-causing genes. The overall goal of the International Porphyria Molecular Diagnostic Collaborative is to determine the pathogenic and benign variants for all eight porphyrias. Here we describe the overall objectives and the initial efforts to validate pathogenic and benign variants in the respective heme biosynthetic genes causing the AHPs.

Entities:  

Keywords:  acute hepatic porphyrias; benign variants; database; pathologic variants; variant validation

Mesh:

Substances:

Year:  2019        PMID: 31073229      PMCID: PMC7229570          DOI: 10.1038/s41436-019-0537-7

Source DB:  PubMed          Journal:  Genet Med        ISSN: 1098-3600            Impact factor:   8.822


  20 in total

1.  General practitioner's radiology casebook IX. Odontome.

Authors:  R E Wood; C J Nortjé
Journal:  J Dent Assoc S Afr       Date:  1988-04

2.  [Emblematic significance of the protective hat in Netherlands literature. Netherlands emblems].

Authors:  H K Hofmeier
Journal:  Med Welt       Date:  1972-10-28

3.  [Editorial: Medical ethics in medical care services].

Authors:  P Kassab
Journal:  AMB Rev Assoc Med Bras       Date:  1973-12

4.  Medical education and medical care. Underserved areas. WAMI: a concept of regionalized medical education.

Authors:  M R Schwarz
Journal:  J Med Educ       Date:  1973-12

5.  [Somatic polysaccharide-containing antigens of the plague bacterium].

Authors:  E E Bakhrakh; V I Veĭnblat
Journal:  Zh Mikrobiol Epidemiol Immunobiol       Date:  1972-03

6.  A 19 F nuclear magnetic resonance study of the binding of trifluoroacetylglucosamine oligomers to lysozyme.

Authors:  F Millett; M A Raftery
Journal:  Biochemistry       Date:  1972-04-25       Impact factor: 3.162

7.  [The possibility of using a glucofuranoside derivative in the treatment of premature lymphedema of the extremities].

Authors:  S Kleczyński
Journal:  Pol Tyg Lek       Date:  1970-06-22

8.  One side of a deletion breakpoint from the Drosophila melanogaster genome contains a transposable element.

Authors:  J Perlman
Journal:  Gene       Date:  1983 Jan-Feb       Impact factor: 3.688

9.  [Beta-endorphin and substance P in the perioperative period].

Authors:  G Krug; P Oehme; M Schädlich
Journal:  Anaesthesiol Reanim       Date:  1991

Review 10.  Porphyrin and heme metabolism and the porphyrias.

Authors:  Herbert L Bonkovsky; Jun-Tao Guo; Weihong Hou; Ting Li; Tarun Narang; Manish Thapar
Journal:  Compr Physiol       Date:  2013-01       Impact factor: 9.090

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  7 in total

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

Authors:  Michael Linenberger; Kleber Y Fertrin
Journal:  Hematology Am Soc Hematol Educ Program       Date:  2020-12-04

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.  Systemic Lupus Erythematosus and Hereditary Coproporphyria: Two Different Entities Diagnosed by WES in the Same Patient.

Authors:  Anlei Liu; Lingli Zhou; Huadong Zhu; Yi Li; Jing Yang
Journal:  Biomed Res Int       Date:  2022-05-28       Impact factor: 3.246

Review 4.  Regulation of Heme Synthesis by Mitochondrial Homeostasis Proteins.

Authors:  Yvette Y Yien; Mark Perfetto
Journal:  Front Cell Dev Biol       Date:  2022-06-27

Review 5.  Porphyrin-Induced Protein Oxidation and Aggregation as a Mechanism of Porphyria-Associated Cell Injury.

Authors:  Dhiman Maitra; Juliana Bragazzi Cunha; Jared S Elenbaas; Herbert L Bonkovsky; Jordan A Shavit; M Bishr Omary
Journal:  Cell Mol Gastroenterol Hepatol       Date:  2019-06-21

Review 6.  Acute Intermittent Porphyria: An Overview of Therapy Developments and Future Perspectives Focusing on Stabilisation of HMBS and Proteostasis Regulators.

Authors:  Helene J Bustad; Juha P Kallio; Marta Vorland; Valeria Fiorentino; Sverre Sandberg; Caroline Schmitt; Aasne K Aarsand; Aurora Martinez
Journal:  Int J Mol Sci       Date:  2021-01-12       Impact factor: 5.923

7.  ABCB6 polymorphisms are not overly represented in patients with porphyria.

Authors:  Colin P Farrell; Gäel Nicolas; Robert J Desnick; Charles J Parker; Jerome Lamoril; Laurent Gouya; Zoubida Karim; Dimitri Tchernitchko; Brenden Chan; Herve Puy; John D Phillips
Journal:  Blood Adv       Date:  2022-02-08
  7 in total

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