Literature DB >> 16150949

Photosensitivity and acute liver injury in myeloproliferative disorder secondary to late-onset protoporphyria caused by deletion of a ferrochelatase gene in hematopoietic cells.

Richard G Goodwin1, W Jonathan Kell, Peter Laidler, C Colin Long, Sharon D Whatley, Mark McKinley, Michael N Badminton, Alan K Burnett, Geraint T Williams, George H Elder.   

Abstract

Late-onset erythropoietic protoporphyria (EPP) is a rare complication of myelodysplastic syndrome (MDS) but has not been described in association with a myeloproliferative disorder (MPD). EPP is normally an inherited disorder characterized by photosensitivity that starts in early childhood and results from overproduction of protoporphyrin secondary to ferrochelatase (FECH) deficiency. Severe liver disease occurs in 1% to 2% of patients. Here we report that severe photosensitivity and cholestatic liver disease in a patient with a myeloproliferative disorder was caused by excess protoporphyrin production from a clone of hematopoietic cells in which one FECH allele had been deleted. Our observations suggest that the usual explanation for the association of late-onset EPP with MPD and MDS is acquired somatic mutation of one FECH allele in bone marrow and show for the first time that the consequent overproduction of protoporphyrin may be severe enough to cause acute liver damage.

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Year:  2005        PMID: 16150949     DOI: 10.1182/blood-2004-12-4939

Source DB:  PubMed          Journal:  Blood        ISSN: 0006-4971            Impact factor:   22.113


  9 in total

Review 1.  [Erythropoietic protoporphyria : Clinical manifestations, diagnosis and new therapeutic possibilities].

Authors:  U Urbanski; J Frank; N J Neumann
Journal:  Hautarzt       Date:  2016-03       Impact factor: 0.751

Review 2.  Erythroid heme biosynthesis and its disorders.

Authors:  Harry A Dailey; Peter N Meissner
Journal:  Cold Spring Harb Perspect Med       Date:  2013-04-01       Impact factor: 6.915

Review 3.  Recent advances on porphyria genetics: Inheritance, penetrance & molecular heterogeneity, including new modifying/causative genes.

Authors:  Makiko Yasuda; Brenden Chen; Robert J Desnick
Journal:  Mol Genet Metab       Date:  2018-11-30       Impact factor: 4.797

4.  Photosensitivity and acute liver insufficiency in late-onset erythropoietic protoporphyria with a chromosome 18q abnormality.

Authors:  Yuka Oshikawa; Satoshi Fukushima; Taiga Miyake; Takeshi Kawaguchi; Kenta Motomura; Yasuhiro Nakashima; Kenichi Nakamura; Masatoshi Jinnin; Hironobu Ihn
Journal:  Case Rep Dermatol       Date:  2012-07-10

Review 5.  Advances in the management of erythropoietic protoporphyria - role of afamelanotide.

Authors:  Ashley M Lane; Jerome T McKay; Herbert L Bonkovsky
Journal:  Appl Clin Genet       Date:  2016-12-12

6.  In vitro Assessment of Solar Filters for Erythropoietic Protoporphyria in the Action Spectrum of Protoporphyrin IX.

Authors:  Alvise Sernicola; Elena Cama; Maria Guglielmina Pelizzo; Enrico Tessarolo; Annamaria Nicolli; Giulia Viero; Mauro Alaibac
Journal:  Front Med (Lausanne)       Date:  2021-12-20

Review 7.  Erythropoietic protoporphyria.

Authors:  Mario Lecha; Hervé Puy; Jean-Charles Deybach
Journal:  Orphanet J Rare Dis       Date:  2009-09-10       Impact factor: 4.123

8.  Anatomic features, tolerance index, secondary metabolites and protein content of chickpea (Cicer arietinum) seedlings under cadmium induction and identification of PCS and FC genes.

Authors:  Elham Mohajel Kazemi; Maryam Kolahi; Milad Yazdi; Andrea Goldson-Barnaby
Journal:  Physiol Mol Biol Plants       Date:  2020-07-29

9.  From the dermatologikum hamburg: quiz.

Authors:  Wendemagegn E Yeshanehe; Rajalakshmi Tirumalae; Almut Böer-Auer
Journal:  Dermatol Pract Concept       Date:  2012-10-31
  9 in total

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