| Literature DB >> 30006060 |
Marion Imbert-Bouteille1, Frédéric Tran Mau Them2, Julien Thevenon3, Thomas Guignard1, Vincent Gatinois1, Jean-Baptiste Riviere4, Anne Boland5, Vincent Meyer5, Jean-François Deleuze5, Elodie Sanchez6, Florence Apparailly7, David Geneviève6, Marjolaine Willems8.
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.Entities:
Keywords: Alazami syndrome; Intellectual disability; LARP7; Primordial dwarfism
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Year: 2018 PMID: 30006060 DOI: 10.1016/j.ejmg.2018.07.003
Source DB: PubMed Journal: Eur J Med Genet ISSN: 1769-7212 Impact factor: 2.708