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LARP7基因变异与原始侏儒症中阿拉扎米综合征表型谱的进一步界定:两姐妹的病例

LARP7 variants and further delineation of the Alazami syndrome phenotypic spectrum among primordial dwarfisms: 2 sisters.

作者信息

Imbert-Bouteille Marion, Mau Them Frédéric Tran, Thevenon Julien, Guignard Thomas, Gatinois Vincent, Riviere Jean-Baptiste, Boland Anne, Meyer Vincent, Deleuze Jean-François, Sanchez Elodie, Apparailly Florence, Geneviève David, Willems Marjolaine

机构信息

Département de Génétique Médicale, Maladies Rares et Médecine Personnalisée, Centre de Référence Anomalies du développement et Syndromes Malformatifs, Plateforme Recherche de Microremaniements Chromosomiques, CHU de Montpellier, Université de Montpellier, France.

Département de Génétique Médicale, Maladies Rares et Médecine Personnalisée, Centre de Référence Anomalies du développement et Syndromes Malformatifs, Plateforme Recherche de Microremaniements Chromosomiques, CHU de Montpellier, Université de Montpellier, France; Unité Inserm, U1183, Hôpital Saint-Eloi, CHU de Montpellier, Montpellier, France; Equipe Génétique des Anomalies du Développement, INSERM UMR1231, Université de Bourgogne-Franche Comté, France; Centre de Génétique et Centre de Référence Anomalies du Développement et Syndromes Malformatifs, FHU TRANSLAD, Hôpital d'Enfants, CHU Dijon et Université de Bourgogne, France.

出版信息

Eur J Med Genet. 2019 Mar;62(3):161-166. doi: 10.1016/j.ejmg.2018.07.003. Epub 2018 Jul 10.

DOI:10.1016/j.ejmg.2018.07.003
PMID:30006060
Abstract

Alazami syndrome (AS) (MIM# 615071) is an autosomal recessive microcephalic primordial dwarfism (PD) with recognizable facial features and severe intellectual disability due to depletion or loss of function variants in LARP7. To date, 15 patients with AS have been reported. Here we describe two consanguineous Algerian sisters with Alazami PD due to LARP7 homozygous pathogenic variants detected by whole exome sequencing. By comparing these two additional cases with those previously reported, we strengthen the key features of AS: severe growth restriction, severe intellectual disability and some distinguishing facial features such as broad nose, malar hypoplasia, wide mouth, full lips and abnormally set teeth. We also report significant new findings enabling further delineation of this syndrome: disproportionately mild microcephaly, stereotypic hand wringing and severe anxiety, thickened skin over the hands and feet, and skeletal, eye and heart malformations. From previous reviews, we summarize the main etiologies of PD according to the involved mechanisms and cellular pathways, highlighting their clinical core features.

摘要

阿拉扎米综合征(AS)(MIM# 615071)是一种常染色体隐性小头原发性侏儒症(PD),因LARP7功能缺失或功能耗竭性变异而具有可识别的面部特征和严重智力残疾。迄今为止,已报道了15例AS患者。在此,我们描述了两名近亲结婚的阿尔及利亚姐妹,她们因通过全外显子测序检测到的LARP7纯合致病变异而患有阿拉扎米型PD。通过将这两例新增病例与先前报道的病例进行比较,我们强化了AS的关键特征:严重生长受限、严重智力残疾以及一些独特的面部特征,如宽鼻、颧骨发育不全、宽嘴、丰满的嘴唇和牙齿排列异常。我们还报告了一些重要的新发现,有助于进一步明确该综合征:相对较轻的小头畸形、刻板的手部扭动和严重焦虑、手脚皮肤增厚以及骨骼、眼睛和心脏畸形。根据以往的综述,我们根据所涉及的机制和细胞途径总结了PD的主要病因,突出了它们的临床核心特征。

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