Literature DB >> 16433813

A Chilean boy with severe photosensitivity and finger shortening: the first case of homozygous variegate porphyria in South America.

P Poblete-Gutiérrez1, C Wolff, R Farias, J Frank.   

Abstract

A 7-year-old Chilean boy presented with severe photosensitivity, blistering, erosions and scarring on sun-exposed areas of the body since the age of 6 months. Additionally, he showed a short stature and shortening of the fingers. Laboratory examination revealed greatly elevated protoporphyrin levels in the blood. Such biochemical findings can be observed in homozygous variants of usually autosomal dominantly inherited acute porphyrias such as variegate porphyria (VP) and hereditary coproporphyria, which usually do not become manifest before the second or third decade of life in heterozygotes. Using polymerase chain reaction-based techniques we identified a missense mutation in exon 7 on the paternal allele and a frameshift mutation in exon 13 on the maternal allele of the protoporphyrinogen oxidase gene that harbours the mutations underlying VP. This is the first homozygous case of VP in South America. As VP represents the most frequent type of acute porphyria not only in Chile but also in South Africa, more such cases could be expected in the future, particularly because a founder mutation for this disease has already been described in the Chilean and South African population.

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Year:  2006        PMID: 16433813     DOI: 10.1111/j.1365-2133.2005.06978.x

Source DB:  PubMed          Journal:  Br J Dermatol        ISSN: 0007-0963            Impact factor:   9.302


  6 in total

1.  Functional Characterization of Five Protoporphyrinogen oxidase Missense Mutations Found in Argentinean Variegate Porphyria Patients.

Authors:  Manuel Méndez; Barbara X Granata; María J Morán Jiménez; Victoria E Parera; Alcira Batlle; Rafael Enríquez de Salamanca; María V Rossetti
Journal:  JIMD Rep       Date:  2011-12-06

2.  Characterization of variegate porphyria mutations using a minigene approach.

Authors:  Barbara Xoana Granata; Marco Baralle; Laura De Conti; Victoria Parera; Maria Victoria Rossetti
Journal:  JIMD Rep       Date:  2015-02-01

Review 3.  [Neurocutaneous porphyrias].

Authors:  J Frank
Journal:  Hautarzt       Date:  2016-03       Impact factor: 0.751

Review 4.  A boy with blistering of sun-exposed skin and finger shortening: the first case of Variegate Porphyria with a novel mutation in protoporphyrinogen oxidase (PPOX) gene in Iran: a case report and literature review.

Authors:  Mohammad Vafaee-Shahi; Saeide Ghasemi; Aina Riahi; Zahra Sadr
Journal:  Ital J Pediatr       Date:  2022-02-14       Impact factor: 2.638

5.  Genetic and biochemical studies in Argentinean patients with variegate porphyria.

Authors:  María V Rossetti; Bárbara X Granata; Jimena Giudice; Victoria E Parera; Alcira Batlle
Journal:  BMC Med Genet       Date:  2008-06-20       Impact factor: 2.103

6.  Porphyria: What Is It and Who Should Be Evaluated?

Authors:  Yonatan Edel; Rivka Mamet
Journal:  Rambam Maimonides Med J       Date:  2018-04-19
  6 in total

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