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一种与GLI-相似3(GLIS3)基因突变相关的新出现的、可识别的面部表型。

An emerging, recognizable facial phenotype in association with mutations in GLI-similar 3 (GLIS3).

作者信息

Dimitri Paul, De Franco Elisa, Habeb Abdelhadi M, Gurbuz Fatih, Moussa Khairya, Taha Doris, Wales Jerry K H, Hogue Jacob, Slavotinek Anne, Shetty Ambika, Balasubramanian Meena

机构信息

Department of Paediatric Endocrinology, Sheffield Children's NHS Foundation Trust, United Kingdom.

Institute of Biomedical and Clinical Science, University of Exeter Medical School, United Kingdom.

出版信息

Am J Med Genet A. 2016 Jul;170(7):1918-23. doi: 10.1002/ajmg.a.37680. Epub 2016 May 5.

Abstract

Neonatal diabetes and hypothyroidism (NDH) syndrome was first described in 2003 in a consanguineous Saudi Arabian family where two out of four siblings were reported to have presented with proportionate IUGR, neonatal non-autoimmune diabetes mellitus, severe congenital hypothyroidism, cholestasis, congenital glaucoma, and polycystic kidneys. Liver disease progressed to hepatic fibrosis. The renal disease was characterized by enlarged kidneys and multiple small cysts with deficient cortico-medullary junction differentiation and normal kidney function. There was minor facial dysmorphism (depressed nasal bridge, large anterior fontanelle, long philtrum) reported but no facial photographs were published. Mutations in the transcription factor GLI-similar 3 (GLIS3) gene in the original family and two other families were subsequently reported in 2006. All affected individuals had neonatal diabetes, congenital hypothyroidism but glaucoma and liver and kidney involvement were less consistent features. Detailed descriptions of the facial dysmorphism have not been reported previously. In this report, we describe the common facial dysmorphism consisting of bilateral low-set ears, depressed nasal bridge with overhanging columella, elongated, upslanted palpebral fissures, persistent long philtrum with a thin vermilion border of the upper lip in a cohort of seven patients with GLIS3 mutations and report the emergence of a distinct, probably recognizable facial gestalt in this group which evolves with age. © 2016 Wiley Periodicals, Inc.

摘要

新生儿糖尿病和甲状腺功能减退症(NDH)综合征于2003年首次在一个沙特阿拉伯近亲家庭中被描述,该家庭的四个兄弟姐妹中有两个被报道出现匀称性宫内生长受限、新生儿非自身免疫性糖尿病、严重先天性甲状腺功能减退症、胆汁淤积、先天性青光眼和多囊肾。肝脏疾病进展为肝纤维化。肾脏疾病的特征是肾脏增大和多个小囊肿,皮质-髓质交界处分化不足,但肾功能正常。有报道称存在轻微面部畸形(鼻梁凹陷、前囟门大、人中长),但未发表面部照片。2006年随后报道了原家庭及其他两个家庭中转录因子GLI-相似3(GLIS3)基因的突变。所有受影响个体均有新生儿糖尿病、先天性甲状腺功能减退症,但青光眼以及肝脏和肾脏受累情况不太一致。此前尚未有对面部畸形的详细描述。在本报告中,我们描述了7例GLIS3基因突变患者中常见的面部畸形,包括双侧低位耳、鼻梁凹陷伴悬垂小柱、睑裂细长且向上倾斜、人中持续较长且上唇红唇缘薄,并报告了该组中一种独特的、可能可识别的面部形态随着年龄增长而演变的情况。© 2016威利期刊公司

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