Your browser doesn't support javascript.
loading
Delineating the phenotype of RNU4ATAC-related spliceosomopathy.
Tabib, Amanda; Richmond, Christopher M; McGaughran, Julie.
Afiliação
  • Tabib A; Paediatrics, John Hunter Children's Hospital, Newcastle, New South Wales, Australia.
  • Richmond CM; Genetic Health QLD, Royal Brisbane & Women's Hospital, Herston, Queensland, Australia.
  • McGaughran J; School of Medicine, Griffith University, Southport, Queensland, Australia.
Am J Med Genet A ; 191(4): 1094-1100, 2023 04.
Article em En | MEDLINE | ID: mdl-36622817
Biallelic pathogenic variants in RNU4ATAC cause microcephalic osteodysplastic primordial dwarfism type I (MOPD1), Roifman syndrome (RS) and Lowry-Wood syndrome (LWS). These conditions demonstrate significant phenotypic heterogeneity yet have overlapping features. Although historically described as discrete conditions they appear to represent a phenotypic spectrum with clinical features not always aligning with diagnostic categories. Clinical variability and ambiguity in diagnostic criteria exist among each disorder. Here we report an individual with a novel genotype and phenotype spanning all three disorders, expanding the phenotypic spectrum of RNU4ATAC-related spliceosomeopathies.
Assuntos
Palavras-chave

Texto completo: 1 Base de dados: MEDLINE Assunto principal: Osteocondrodisplasias / Nanismo / Microcefalia Limite: Female / Humans Idioma: En Ano de publicação: 2023 Tipo de documento: Article

Texto completo: 1 Base de dados: MEDLINE Assunto principal: Osteocondrodisplasias / Nanismo / Microcefalia Limite: Female / Humans Idioma: En Ano de publicação: 2023 Tipo de documento: Article