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Expanding the clinical and molecular spectrum of the CWC27-related spliceosomopathy.
Brea-Fernández, Alejandro J; Cabanas, Paloma; Dacruz-Álvarez, David; Caamaño, Pilar; Limeres, Jacobo; Loidi, Lourdes.
Afiliação
  • Brea-Fernández AJ; Grupo de Medicina Xenómica, Universidad de Santiago de Compostela, Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER), Santiago de Compostela, Spain. a.brea@usc.es.
  • Cabanas P; Complexo Hospitalario Universitario de Santiago de Compostela, Unidad de Endocrinología Pediátrica, Departamento de Pediatría, Santiago de Compostela, Spain.
  • Dacruz-Álvarez D; Complexo Hospitalario Universitario de Santiago de Compostela, Unidad de Neurología Pediátrica, Departamento de Pediatría, Santiago de Compostela, Spain.
  • Caamaño P; Fundación Pública Galega de Medicina Xenómica-SERGAS, Santiago de Compostela, Spain.
  • Limeres J; Unidad de Odontología para Personas con Necesidades Especiales, Facultad de Medicina y Odontología, Universidad de Santiago de Compostela, Santiago de Compostela, Spain.
  • Loidi L; Fundación Pública Galega de Medicina Xenómica-SERGAS, Santiago de Compostela, Spain.
J Hum Genet ; 64(11): 1133-1136, 2019 Nov.
Article em En | MEDLINE | ID: mdl-31481716
Cyclophilins are a type of peptidyl-prolyl cis-trans isomerases. CWC27, one of the known human cyclophilins, is recruited by the spliceosome for the pre-mRNA splicing process. Biallelic deleterious variants in CWC27 lead to a spectrum of overlapping phenotypes including retinal degeneration, skeletal anomalies, short stature, and neurological defects. The present work reports a woman showing these clinical features, in addition to hypergonadotropic hypogonadism, hypoplastic/agenesic teeth, and cataracts, not previously associated with such phenotypic spectrum. Whole exome sequencing on this patient identified a novel CWC27 homozygous variant predicted to originate a severely truncated protein and the consequent loss of functionality. The clinical and genetic characterization of such patient could provide further insight into the underlying causes of the spliceosomopathies.
Assuntos

Texto completo: 1 Bases de dados: MEDLINE Assunto principal: Anormalidades Múltiplas / Ciclofilinas / Sequenciamento do Exoma Tipo de estudo: Prognostic_studies Limite: Female / Humans / Infant Idioma: En Revista: J Hum Genet Assunto da revista: GENETICA MEDICA Ano de publicação: 2019 Tipo de documento: Article País de afiliação: Espanha

Texto completo: 1 Bases de dados: MEDLINE Assunto principal: Anormalidades Múltiplas / Ciclofilinas / Sequenciamento do Exoma Tipo de estudo: Prognostic_studies Limite: Female / Humans / Infant Idioma: En Revista: J Hum Genet Assunto da revista: GENETICA MEDICA Ano de publicação: 2019 Tipo de documento: Article País de afiliação: Espanha