Delineating the phenotype of RNU4ATAC-related spliceosomopathy.
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.
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Texto completo:
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Base de dados:
MEDLINE
Assunto principal:
Osteocondrodisplasias
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Nanismo
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Microcefalia
Limite:
Female
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Humans
Idioma:
En
Ano de publicação:
2023
Tipo de documento:
Article