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Lessons of a day hospital: Comprehensive assessment of patients with albinism in a European setting.
Marti, Aurélie; Lasseaux, Eulalie; Ezzedine, Khaled; Léauté-Labrèze, Christine; Boralevi, Franck; Paya, Clément; Coste, Valentine; Deroissart, Vincent; Arveiler, Benoit; Taieb, Alain; Morice-Picard, Fanny.
Afiliação
  • Marti A; Paediatric Dermatology Unit, National Reference Center for Rare Skin Disorders, Hôpital Pellegrin-Enfants, Bordeaux University Hospitals, Bordeaux, France.
  • Lasseaux E; Molecular Genetics Laboratory, CHU de Bordeaux, Bordeaux, France.
  • Ezzedine K; Department of Dermatology, AP-HP, Hôpital Henri-Mondor, Créteil, France.
  • Léauté-Labrèze C; Paediatric Dermatology Unit, National Reference Center for Rare Skin Disorders, Hôpital Pellegrin-Enfants, Bordeaux University Hospitals, Bordeaux, France.
  • Boralevi F; Paediatric Dermatology Unit, National Reference Center for Rare Skin Disorders, Hôpital Pellegrin-Enfants, Bordeaux University Hospitals, Bordeaux, France.
  • Paya C; Ophthalmology Department, CHU de Bordeaux, Bordeaux, France.
  • Coste V; Ophthalmology Department, CHU de Bordeaux, Bordeaux, France.
  • Deroissart V; Institute of Public Health, Epidemiology and Development "ISPED", Bordeaux University, Bordeaux, France.
  • Arveiler B; Molecular Genetics Laboratory, CHU de Bordeaux, Bordeaux, France.
  • Taieb A; Unité INSERM U1211, Maladies Rares: Génétique et Métabolisme, Bordeaux, France.
  • Morice-Picard F; Paediatric Dermatology Unit, National Reference Center for Rare Skin Disorders, Hôpital Pellegrin-Enfants, Bordeaux University Hospitals, Bordeaux, France.
Pigment Cell Melanoma Res ; 31(2): 318-329, 2018 03.
Article em En | MEDLINE | ID: mdl-28976636
Albinism is a rare genetic disease, comprising syndromic and non-syndromic forms. We assessed clinical and genetic characteristics in a prospective evaluation of 64 patients (33 children and 31 adults) seen at a specialized day hospital. Causative genetic mutations were found in TYR (23/64, 35.9%), OCA2 (19/64, 29.7%), TYRP1 (1/64, 1.6%), SLC45A2 (12/64, 18.7%), C10orf11 (1/64, 1.6%), HPS1 (3/64, 4.7%), HPS5 (1/64, 1.5%), HPS6 (1/64, 1.6%) and GPR143 (2/64, 3.1%). Causative mutations remained undetermined for one patient (1.6%). Heterogeneity for hair and skin phenotype was noted across and within the different genotypes. Skin and hair hypopigmentation did not correlate with visual impairment. The diagnosis of unrecognized syndromic forms and of cases of ocular albinism in this prospective and comprehensive series of patients with albinism in a European setting is remarkable. Photoprotection was overall good but not optimal.
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Texto completo: 1 Base de dados: MEDLINE Assunto principal: Albinismo Idioma: En Ano de publicação: 2018 Tipo de documento: Article

Texto completo: 1 Base de dados: MEDLINE Assunto principal: Albinismo Idioma: En Ano de publicação: 2018 Tipo de documento: Article