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Analysis of COL7A1 pathogenic variants in a large cohort of dystrophic epidermolysis bullosa patients from Argentina reveals a new genotype-phenotype correlation.
Natale, Mónica Inés; Manzur, Graciela Beatriz; Lusso, Silvina Beatriz; Cella, Eliana; Giovo, María Elsa; Andrada, Romina; Goitia, Juana; Fernández, María Florencia; Della Giovanna, Patricia Silvia; Guillamondegui, María José; Domínguez, Mariángeles; Gutiérrez, Olga; Izquierdo, Agustín; Hernández Herrera, Heliana; Velázquez Perdomo, Luz Graciela; Mistchenko, Alicia Susana; Valinotto, Laura Elena.
Afiliación
  • Natale MI; Center for Research in Genodermatoses and Epidermolysis Bullosa (CEDIGEA), University of Buenos Aires, Buenos Aires, Argentina.
  • Manzur GB; Center for Research in Genodermatoses and Epidermolysis Bullosa (CEDIGEA), University of Buenos Aires, Buenos Aires, Argentina.
  • Lusso SB; Rare Diseases of the Skin Unit, Dr. R. Gutierrez Children's Hospital, Buenos Aires, Argentina.
  • Cella E; Dermatology Department, Hospital de Clinicas "Jose de San Martín", Buenos Aires, Argentina.
  • Giovo ME; Center for Research in Genodermatoses and Epidermolysis Bullosa (CEDIGEA), University of Buenos Aires, Buenos Aires, Argentina.
  • Andrada R; Pediatric Dermatology, Prof. Dr. Juan P. Garrahan Children's Hospital, Buenos Aires, Argentina.
  • Goitia J; Pediatric Dermatology, La Santisima Trinidad Children's Hospital, Córdoba, Argentina.
  • Fernández MF; Dermatology, Avelino Castelan Children's Hospital, Resistencia, Chaco, Argentina.
  • Della Giovanna PS; Pediatric Dermatology, Sor Maria Ludovica Children's Hospital, La Plata, Buenos Aires, Argentina.
  • Guillamondegui MJ; Pediatric Dermatology, Fundacion Respirar, Argentina.
  • Domínguez M; Dermatology, Prof. A. Posadas Hospital, El Palomar, Buenos Aires, Argentina.
  • Gutiérrez O; Genetics Unit, Humberto J. Notti Children's Hospital, Guaymallen, Mendoza, Argentina.
  • Izquierdo A; Pediatric Dermatology, Hospital General de Agudos "Carlos G. Durand", Buenos Aires, Argentina.
  • Hernández Herrera H; Pediatric Dermatology, Niños de Acosta Ñu Children's Hospital, San Lorenzo, Paraguay.
  • Velázquez Perdomo LG; Bioinformatics, Translational Research Unit, Dr. R. Gutiérrez Children's Hospital, Buenos Aires, Argentina.
  • Mistchenko AS; Center for Research in Genodermatoses and Epidermolysis Bullosa (CEDIGEA), University of Buenos Aires, Buenos Aires, Argentina.
  • Valinotto LE; Dermatology Department, Hospital de Clinicas "Jose de San Martín", Buenos Aires, Argentina.
Am J Med Genet A ; 188(11): 3153-3161, 2022 11.
Article en En | MEDLINE | ID: mdl-35979658
Dystrophic epidermolysis bullosa (DEB) is a clinically heterogeneous heritable skin disorder, characterized by blistering of the skin and mucous membranes following minor trauma. Dominant (DDEB) and recessive (RDEB) forms are caused by pathogenic variants in COL7A1 gene. Argentina's population has a heterogeneous genetic background, and little is known about the molecular basis of DEB in our country or in native South American populations. In this study, we present the prevalence and geographical distribution of pathogenic variants found in 181 patients from 136 unrelated families (31 DDEB and 105 RDEB). We detected 95 different variants, 59 of them were previously reported in the literature and 36 were novel, nine of which were detected in more than one family. The most prevalent pathogenic variants were identified in exon 73 in DDEB patients and in exon 3 in RDEB patients. We also report a new phenotype-genotype correlation found in 10 unrelated families presenting mild blistering and severe mucosal involvement. Molecular studies in populations with an unexplored genetic background like ours revealed a diversity of pathogenic variants, and we hope that these findings will contribute to the definition of targets for new gene therapies.
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Texto completo: 1 Colección: 01-internacional Banco de datos: MEDLINE Asunto principal: Epidermólisis Ampollosa Distrófica / Colágeno Tipo VII Tipo de estudio: Prognostic_studies / Risk_factors_studies Límite: Humans País/Región como asunto: America do sul / Argentina Idioma: En Revista: Am J Med Genet A Asunto de la revista: GENETICA MEDICA Año: 2022 Tipo del documento: Article País de afiliación: Argentina

Texto completo: 1 Colección: 01-internacional Banco de datos: MEDLINE Asunto principal: Epidermólisis Ampollosa Distrófica / Colágeno Tipo VII Tipo de estudio: Prognostic_studies / Risk_factors_studies Límite: Humans País/Región como asunto: America do sul / Argentina Idioma: En Revista: Am J Med Genet A Asunto de la revista: GENETICA MEDICA Año: 2022 Tipo del documento: Article País de afiliación: Argentina