Literature DB >> 27544718

A novel splice site mutation of FGD1 gene in an Aarskog-Scott syndrome patient with a large anterior fontanel.

Erhan Parıltay, Filiz Hazan, Esra Ataman, Korcan Demir, Özdal Etlik, Erhan Özbek, Behzat Özkan.   

Abstract

Aarskog-Scott syndrome (ASS) is a rare X-linked recessive genetic disorder caused by FGD1 mutations. FGD1 regulates the actin cytoskeleton and regulates cell growth and differentiation by activating the c-Jun N-terminal kinase signaling cascade. ASS is characterized by craniofacial dysmorphism, short stature, interdigital webbing and shawl scrotum. However, there is a wide phenotypic heterogeneity because of the additional clinical features. ASS and some syndromes including the autosomal dominant inherited form of Robinow syndrome, Noonan syndrome, pseudohypoparathyroidism, Silver-Russel and SHORT syndrome have some overlapping phenotypic features. Herein, we report a patient with ASS and a large anterior fontanel who was initially diagnosed as Robinow syndrome. He was found to have a novel c.1340+2 T>A splice site mutation on the FGD1 gene.

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Year:  2016        PMID: 27544718     DOI: 10.1515/jpem-2015-0482

Source DB:  PubMed          Journal:  J Pediatr Endocrinol Metab        ISSN: 0334-018X            Impact factor:   1.634


  2 in total

Review 1.  The Prevalence of Clinical Features in Patients with Aarskog-Scott Syndrome and Assessment of Genotype-Phenotype Correlation: A Systematic Review.

Authors:  Victor Zanetti Drumond; Lucas Sousa Salgado; Camila Sousa Salgado; Vitor Augusto de Lima Oliveira; Eliene Magda de Assis; Michel Campos Ribeiro; Analina Furtado Valadão; Alfredo Orrico
Journal:  Genet Res (Camb)       Date:  2021-02-02       Impact factor: 1.588

2.  A novel frameshift mutation in the FGD1 gene causing Aarskog-Scott syndrome patient with hypogonadism: a case report.

Authors:  Hongshuai Jia; Tiantian Ma; Ziqin Liu; Yuru Ouyang; Chunsheng Hao
Journal:  Transl Pediatr       Date:  2021-05
  2 in total

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