Literature DB >> 15241683

Variable effects of beta-carotene therapy in a child with erythropoietic protoporphyria.

Ramin Alemzadeh1, Tammy Feehan.   

Abstract

UNLABELLED: Erythropoietic protoporphyria (EPP) is an inborn error of heme biosynthesis with high levels of protoporphyrin in red cells and is characterized by mild to moderate photosensitivity. High-dose beta-carotene therapy has been reported to afford photoprotection in patients with EPP. We report the case of a 5-year-old Caucasian female with EPP who presented with a long-standing 3-year history of recurrent facial blisters and erythematous swelling and lesions of other sun-exposed areas of the skin. She was treated with a topical sunblocker (PreSun Ultra-SP45) but continued to show moderate to severe photosensitivity despite 3 months (March to May) of vigorous photoprotection. She was then started on increasing doses of beta-carotene (90-180 mg/day) over a period of 3 months (June to September) which resulted in a marked improvement of both facial and forearm lesions, but only modest improvement in her hand lesions.
CONCLUSION: High-dose beta-carotene appears to provide photoprotection in erythropoietic protoporphyria, resulting in improved but highly variable tolerance to sunlight.

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Year:  2004        PMID: 15241683     DOI: 10.1007/s00431-004-1453-6

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  12 in total

1.  Photodynamic effects of protoporphyrin on red blood cell deformability.

Authors:  T M Dubbelman; A W de Bruijne; J van Steveninck
Journal:  Biochem Biophys Res Commun       Date:  1977-08-08       Impact factor: 3.575

2.  Singlet oxygen as a mediator in the hematoporphyrin-catalyzed photooxidation of NADPH to NADP+ in deuterium oxide.

Authors:  R S Bodaness; P C Chan
Journal:  J Biol Chem       Date:  1977-12-10       Impact factor: 5.157

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Authors:  M M Mathews-Roth
Journal:  Photochem Photobiol       Date:  1986-01       Impact factor: 3.421

4.  Porphyrin-induced photodamage at the cellular and the subcellular level as related to the solubility of the porphyrin.

Authors:  S Sandberg; I Romslo
Journal:  Clin Chim Acta       Date:  1981-01-22       Impact factor: 3.786

5.  Genetic heterogeneity in erythropoietic protoporphyria: a study of the enzymatic defect in nine affected families.

Authors:  P G Norris; A V Nunn; J L Hawk; T M Cox
Journal:  J Invest Dermatol       Date:  1990-09       Impact factor: 8.551

6.  An unusual case of purpuric erythropoietic protoporphyria.

Authors:  G K Patel; J Weston; E K Derrick; J L Hawk
Journal:  Clin Exp Dermatol       Date:  2000-07       Impact factor: 3.470

7.  The histopathology and ultrastructure of liver disease in erythropoietic protoporphyria.

Authors:  D M MacDonald; D Germain; H Perrot
Journal:  Br J Dermatol       Date:  1981-01       Impact factor: 9.302

Review 8.  Erythropoietic protoporphyria.

Authors:  D J Todd
Journal:  Br J Dermatol       Date:  1994-12       Impact factor: 9.302

9.  Erythropoietic protoporphyria: lipid peroxidation and red cell membrane damage associated with photohemolysis.

Authors:  B D Goldstein; L C Harber
Journal:  J Clin Invest       Date:  1972-04       Impact factor: 14.808

10.  Beta-carotene in erythropoietic protoporphyria: 5 years' experience.

Authors:  K Thomsen; H Schmidt; A Fischer
Journal:  Dermatologica       Date:  1979
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  4 in total

Review 1.  Identification of FECH gene multiple variations in two Chinese patients with erythropoietic protoporphyria and a review.

Authors:  Zhang-Biao Long; Yong-Wei Wang; Chen Yang; Gang Liu; Ya-Li Du; Guang-Jun Nie; Yan-Zhong Chang; Bing Han
Journal:  J Zhejiang Univ Sci B       Date:  2016 Oct.       Impact factor: 3.066

Review 2.  Nutrients and Porphyria: An Intriguing Crosstalk.

Authors:  Elena Di Pierro; Francesca Granata
Journal:  Int J Mol Sci       Date:  2020-05-14       Impact factor: 5.923

3.  Erythropoietic protoporphyria without skin symptoms-you do not always see what they feel.

Authors:  Anne L Y Lecluse; Veronica C M Kuck-Koot; Huib van Weelden; Vigfus Sigurdsson; Ingrid M Russel; Jorge Frank; Suzanne G M A Pasmans
Journal:  Eur J Pediatr       Date:  2007-08-21       Impact factor: 3.183

4.  Protective action of antioxidants on hepatic damage induced by griseofulvin.

Authors:  M del C Martinez; S G Afonso; A M Buzaleh; A Batlle
Journal:  ScientificWorldJournal       Date:  2014-01-12
  4 in total

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