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A novel deletion mutation in the ATP6V0A2 gene in an Iranian patient affected by autosomal recessive cutis laxa.
Shafagh Shishavan, Negar; Morovvati, Saeid.
Afiliação
  • Shafagh Shishavan N; Department of Genetics, Faculty of Advanced Sciences and Technology, Tehran Medical Sciences, Islamic Azad University, Tehran, Iran.
  • Morovvati S; Department of Biology, Faculty of Sciences, University of Guilan, Rasht, Iran.
Ir J Med Sci ; 192(5): 2279-2282, 2023 Oct.
Article em En | MEDLINE | ID: mdl-36520350
Cutis laxa (CL) can be caused by mutations in a number of genes. Cutis laxa with autosomal recessive inheritance due to mutations in several genes, including mutations in the ATP6V0A2 gene, causes autosomal recessive cutis laxa type 2A (ARCL2A). The ATP6V0A2 gene encodes the a2 subunit in the V-ATPases pump. The V-ATPases are located in the membrane of some organelles, including the Golgi or some vesicles, and act as ATP-dependent proton pumps to pH adjustment intracellular segments. Mutations in the ATP6V0A2 gene consist present in ARCL2A patients. We present the case of a 12-year-old girl who was referred to Rasad Laboratory (Tehran, Iran) at the age of 5 with a set of symptoms of congenital disorders. Her clinical phenotype contains distal symmetrical sensory and motor polyneuropathy, loose joints, large nasal roots, growth delay, and wrinkled skin. Also, there was a history of the parental marriage of consanguinity. A potentially pathogenic homozygous deletion mutation was detected in the ATP6V0A2 gene related to ARCL2A. This mutation has not been reported in the other patients with ARCL2A. A novel homozygous deletion mutation in ATP6V0A2 is supposed to be the reason for disease in our proband.
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Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Cútis Laxa Limite: Child / Female / Humans País/Região como assunto: Asia Idioma: En Revista: Ir J Med Sci Ano de publicação: 2023 Tipo de documento: Article

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Cútis Laxa Limite: Child / Female / Humans País/Região como assunto: Asia Idioma: En Revista: Ir J Med Sci Ano de publicação: 2023 Tipo de documento: Article