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DOI 10.1002/ajmg.a.62706

Медицинская библиотека · American journal of medical genetics. Part A · 2022

BRPF1-associated syndrome: A patient with congenital ptosis, neurological findings, and normal intellectual development.

Souza Josiane, do Valle Daniel Almeida, Santos Mara Lucia Schmidt Ferreira, Colomé Fernanda Bonilla, Teive Helio Afonso Ghizoni, da Silva Freitas Renato, Herai Roberto Hirochi

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Аннотация

In 2017, Mattiolli et al. and Yan et al. described a series of patients with clinical findings essentially characterized by intellectual disabilities, ptosis, hypotonia, epilepsy, and weakness. They also found in these patients distinct heterozygous mutations in the BRPF1 gene, which plays a role in epigenetic regulation by promoting histone acetylation. The disease is known as Intellectual Developmental Disorder with Dysmorphic Facies and Ptosis (IDDDFP, OMIM #617333). Later, another 20 patients were also described by distinct reports, suggesting IDDDFP could be a more frequent cause of intellectual disability as it was thought before. Here, we describe a patient with normal intellectual development who had congenital ptosis, hypotonia, muscular weakness, atlanto-axial malformation, and pyramidal at the neurological examination. The patient has a rare nonsense variant on exon 3 of BRPF1 gene. We also describe a phenotypic amplification for conditions related to deficiency in histone modifications.

Для цитирования: Souza Josiane, do Valle Daniel Almeida, Santos Mara Lucia Schmidt Ferreira, Colomé Fernanda Bonilla, Teive Helio Afonso Ghizoni, da Silva Freitas Renato, Herai Roberto Hirochi. BRPF1-associated syndrome: A patient with congenital ptosis, neurological findings, and normal intellectual development.. American journal of medical genetics. Part A. 2022. DOI: 10.1002/ajmg.a.62706.

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