Literature DB >> 8404971

Ocular involvement in two symptomatic congenital erythropoietic porphyria.

F Oguz1, M Sidal, C Bayram, N Sansoy, N Hekim.   

Abstract

Congenital erythropoietic porphyria (Gunther disease, CEP) is a rare autosomal recessive disorder of haeme biosynthesis. It is characterized by extreme photosensitivity and the excretion of large amounts of uroporphyrin I and coproporphyrin I in the urine and coproporphyrin I in the faeces. We have diagnosed two cases of congenital erythropoietic porphyria, who were first cousins once removed. They had recurrent skin bullae, scarring on the face and hands, hirsutism, discoloured fluorescent teeth, red urine, increased haemolysis and grossly increased excretion of porphyrin. Both children had blepharitis and their sclera gave pink fluorescence under long wave ultraviolet light, mainly in the interpalpebral fissures. All the features of our two patients, except the ocular lesions, conformed to cases of CEP reported in the literature. We have encountered no other reports on ocular lesions in CEP since first described by Chumbley in 1977.

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Year:  1993        PMID: 8404971     DOI: 10.1007/bf01955245

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


  7 in total

1.  Some studies of the comparative biology of human and bovine porphyria erythropoietica.

Authors:  C J WATSON; V PERMAN; F A SPURRELL; H H HOYT; S SCHWARTZ
Journal:  Trans Assoc Am Physicians       Date:  1958

2.  Scleral involvement in symptomatic porphyria.

Authors:  L C Chumbley
Journal:  Am J Ophthalmol       Date:  1977-11       Impact factor: 5.258

3.  Congenital erythropoietic porphyria. An autopsy report.

Authors:  L K Bhutani; S K Sood; P K Das; D N Mulay; K C Kandhari; S G Deshpande
Journal:  Arch Dermatol       Date:  1974-09

4.  Congenital erythropoietic porphyria. I. Case report, special studies and therapy.

Authors:  R G Haining; M L Cowger; D B Shurtleff; R F Labbe
Journal:  Am J Med       Date:  1968-10       Impact factor: 4.965

5.  Prenatal exclusion of congenital erythropoietic porphyria (Günther's disease) in a fetus at risk.

Authors:  J C Deybach; B Grandchamp; M Grelier; Y Nordmann; J Boué; A Boué; P de Berrianger
Journal:  Hum Genet       Date:  1980-02       Impact factor: 4.132

6.  Point mutations in the uroporphyrinogen III synthase gene in congenital erythropoietic porphyria (Günther's disease).

Authors:  J C Deybach; H de Verneuil; S Boulechfar; B Grandchamp; Y Nordmann
Journal:  Blood       Date:  1990-05-01       Impact factor: 22.113

7.  Congenital erythropoietic porphyria (Günther's disease): enzymatic studies on two cases of late onset.

Authors:  J C Deybach; H de Verneuil; N Phung; Y Nordmann; A Puissant; B Boffety
Journal:  J Lab Clin Med       Date:  1981-04
  7 in total
  2 in total

Review 1.  Congenital erythropoietic porphyria: Recent advances.

Authors:  Angelika L Erwin; Robert J Desnick
Journal:  Mol Genet Metab       Date:  2018-12-27       Impact factor: 4.797

2.  Scleral necrosis in congenital erythropoietic porphyria: A case report and review of the literature.

Authors:  Shweta Agarwal; Parthopratim Dutta Majumder; Bhaskar Srinivasan; Geetha Iyer
Journal:  Oman J Ophthalmol       Date:  2015 Sep-Dec
  2 in total

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