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DOI 10.1002/humu.23980

Медицинская библиотека · Human mutation · 2020

Genomics-based treatment in a patient with two overlapping heritable skin disorders: Epidermolysis bullosa and acrodermatitis enteropathica.

Vahidnezhad Hassan, Youssefian Leila, Sotoudeh Soheila, Liu Lu, Guy Alyson, Lovell Patricia A, Kariminejad Ariana, Zeinali Sirous, McGrath John A, Uitto Jouni

Клинический случайСтатьяПоддержка исследования: вне правительства СШАПеревод в очереди

Аннотация

Next-generation sequencing (NGS) is helpful in diagnosing complex genetic disorders and phenotypes, particularly when more than one overlapping condition is present. From a large cohort of 362 families with clinical manifestations of skin and mucosal fragility, referred by several major medical centers, one patient was found by NGS to have two overlapping heritable skin diseases, recessive dystrophic epidermolysis bullosa (RDEB; COL7A1 mutations) and acrodermatitis enteropathica (AE; SLC39A4 mutations). The pathogenicity of the variants was studied at gene expression as well as ultrastructural and tissue levels. Although there is no specific treatment for RDEB except avoiding trauma, supplementation with oral zinc (3 mg·kg-1 ·day-1 ) for the AE resulted in rapid amelioration of the skin findings. This case demonstrates the power of NGS in identifying two genetically unlinked diseases that led to effective treatment with major clinical benefits as an example of genomics-guided treatment.

Для цитирования: Vahidnezhad Hassan, Youssefian Leila, Sotoudeh Soheila, Liu Lu, Guy Alyson, Lovell Patricia A, Kariminejad Ariana, Zeinali Sirous, McGrath John A, Uitto Jouni. Genomics-based treatment in a patient with two overlapping heritable skin disorders: Epidermolysis bullosa and acrodermatitis enteropathica.. Human mutation. 2020. DOI: 10.1002/humu.23980.

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