Frontiers in pediatrics · 2023
Case Report: Novel biallelic moderately damaging variants in RTTN in a patient with cerebellar dysplasia.
СтатьяНазвание и аннотация: Перевод в очереди
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Romano Ferruccio, Amadori Elisabetta, Madia Francesca, Severino Mariasavina, Capra Valeria, Rizzo Renata, Barone Rita, Corradi Beatrice, Maragliano Luca, Shams Nosrati Mohammad Sadegh, Falace Antonio, Striano Pasquale, Zara Federico, Scala Marcello
Аннотация
Rotatin, encoded by the RTTN gene, is a centrosomal protein with multiple, emerging functions, including left-right specification, ciliogenesis, and neuronal migration. Recessive variants in RTTN are associated with a neurodevelopmental disorder with microcephaly and malformations of cortical development known as "Microcephaly, short stature, and polymicrogyria with seizures" (MSSP, MIM #614833). Affected individuals show a wide spectrum of clinical manifestations like intellectual disability, poor/absent speech, short stature, microcephaly, and congenital malformations. Here, we report a subject showing a distinctive neuroradiological phenotype and harboring novel biallelic variants in RTTN: the c.5500A>G, p.(Asn1834Asp), (dbSNP: rs200169343, ClinVar ID:1438510) and c.19A>G, p.(Ile7Val), (dbSNP: rs201165599, ClinVar ID:1905275) variants. In particular brain magnetic resonance imaging (MRI) showed a peculiar pattern, with cerebellar hypo-dysplasia, and multiple arachnoid cysts in the lateral cerebello-medullary cisterns, in addition to left Meckel cave. Thus, we compare his phenotypic features with current literature, speculating a possible role of newly identified RTTN variants in his clinical picture, and supporting a relevant variability in this emerging condition.