Literature DB >> 1251847

Erythropoietic protoporphyria. 10 years experience.

V A DeLeo, M Poh-Fitzpatrick, M Mathews-Roth, L C Harber.   

Abstract

The clinical and laboratory findings in 32 patients with erythropoietic protoporphyria as well as a review of the pertinent literature on this relatively recently described form of porphyria are presented. The disease is thought to be transmitted in an autosomal dominant fashion with variable penetrance and was characterized in these 32 patients by the onset in childhood of burning (97 per cent) and itching (88 per cent) of the skin on exposure to sunlight. This was accompanied by edema (49 per cent) and erythema (69 per cent) of the exposed areas. Vesicles, petechiae and residual scarring occurred less frequently. Associated abnormalities included cholelithiasis (12 per cent), anemia (27 per cent) and abnormal liver function studies (4 per cent). Reports of associated liver disease including nine cases of fatal hepatic failure, are reviewed. Current methods of diagnosis as well as theories of pathophysiology of the disease are presented. Nineteen of 23 of these patients recently treated with beta-carotene responded with significant increase in their tolerance to sun exposure.

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Year:  1976        PMID: 1251847     DOI: 10.1016/0002-9343(76)90528-3

Source DB:  PubMed          Journal:  Am J Med        ISSN: 0002-9343            Impact factor:   4.965


  27 in total

1.  The diagnosis and management of erythropoietic protoporphyria.

Authors:  Manish Thapar; Herbert L Bonkovsky
Journal:  Gastroenterol Hepatol (N Y)       Date:  2008-08

Review 2.  Liver disease in erythropoietic protoporphyria: insights and implications for management.

Authors:  A V Anstey; R J Hift
Journal:  Postgrad Med J       Date:  2007-12       Impact factor: 2.401

Review 3.  Liver disease in erythropoietic protoporphyria: insights and implications for management.

Authors:  A V Anstey; R J Hift
Journal:  Gut       Date:  2007-03-14       Impact factor: 23.059

4.  The molecular defect of ferrochelatase in a patient with erythropoietic protoporphyria.

Authors:  Y Nakahashi; H Fujita; S Taketani; N Ishida; A Kappas; S Sassa
Journal:  Proc Natl Acad Sci U S A       Date:  1992-01-01       Impact factor: 11.205

Review 5.  Erythropoietic protoporphyria.

Authors:  T M Cox
Journal:  J Inherit Metab Dis       Date:  1997-06       Impact factor: 4.982

6.  Study of factors causing excess protoporphyrin accumulation in cultured skin fibroblasts from patients with protoporphyria.

Authors:  J R Bloomer; D A Brenner; M J Mahoney
Journal:  J Clin Invest       Date:  1977-12       Impact factor: 14.808

Review 7.  Progress in the field of fat-soluble vitamins and carotenoids.

Authors:  O Isler
Journal:  Experientia       Date:  1977-05-15

8.  Microcytic anemia, erythropoietic protoporphyria, and neurodegeneration in mice with targeted deletion of iron-regulatory protein 2.

Authors:  Sharon S Cooperman; Esther G Meyron-Holtz; Hayden Olivierre-Wilson; Manik C Ghosh; Joseph P McConnell; Tracey A Rouault
Journal:  Blood       Date:  2005-04-14       Impact factor: 22.113

9.  A second attack of cholestasis associated with erythropoietic protoporphyria was successfully treated by plasma exchange and blood transfusion.

Authors:  Yohji Honda; Yoshiiku Kawakami; Hiromi Kan; Hatsue Fujino; Takayuki Fukuhara; Noriaki Naeshiro; Daisuke Miyaki; Tomokazu Kawaoka; Akira Hiramatsu; Masataka Tsuge; Michio Imamura; Hideyuki Hyogo; Hiroshi Aikata; Kazuaki Chayama
Journal:  Clin J Gastroenterol       Date:  2014-06-10

10.  Erythropoietic protoporphyria in the house mouse. A recessive inherited ferrochelatase deficiency with anemia, photosensitivity, and liver disease.

Authors:  S Tutois; X Montagutelli; V Da Silva; H Jouault; P Rouyer-Fessard; K Leroy-Viard; J L Guénet; Y Nordmann; Y Beuzard; J C Deybach
Journal:  J Clin Invest       Date:  1991-11       Impact factor: 14.808

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