Literature DB >> 30006060

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

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.
Copyright © 2018 Elsevier Masson SAS. All rights reserved.

Entities:  

Keywords:  Alazami syndrome; Intellectual disability; LARP7; Primordial dwarfism

Mesh:

Substances:

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


  5 in total

1.  Stabilize and connect: the role of LARP7 in nuclear non-coding RNA metabolism.

Authors:  Daniele Hasler; Gunter Meister; Utz Fischer
Journal:  RNA Biol       Date:  2020-06-03       Impact factor: 4.652

2.  Updating the neurodevelopmental profile of Alazami syndrome: Illustrating the role of developmental assessment in rare genetic disorders.

Authors:  Monica H Wojcik; Kate Linnea; Joan M Stoler; Leonard Rappaport
Journal:  Am J Med Genet A       Date:  2019-05-10       Impact factor: 2.802

3.  de novo MEPCE nonsense variant associated with a neurodevelopmental disorder causes disintegration of 7SK snRNP and enhanced RNA polymerase II activation.

Authors:  Pauline E Schneeberger; Tatjana Bierhals; Axel Neu; Maja Hempel; Kerstin Kutsche
Journal:  Sci Rep       Date:  2019-08-29       Impact factor: 4.379

4.  Compound Phenotype Due to Recessive Variants in LARP7 and OTOG Genes Disclosed by an Integrated Approach of SNP-Array and Whole Exome Sequencing.

Authors:  Pietro Palumbo; Orazio Palumbo; Maria Pia Leone; Ester di Muro; Stefano Castellana; Luigi Bisceglia; Tommaso Mazza; Massimo Carella; Marco Castori
Journal:  Genes (Basel)       Date:  2020-03-31       Impact factor: 4.096

5.  Patient with Phenylketonuria and Intellectual Disability-Problem Not Always Caused Exclusively by Insufficient Metabolic Control (Coexistence of PKU and Alazami Syndrome).

Authors:  Michał Patalan; Alicja Leśniak; Krzysztof Bernatowicz; Hanna Romanowska; Elżbieta Krzywińska-Zdeb; Mieczysław Walczak; Maria Giżewska
Journal:  Int J Environ Res Public Health       Date:  2022-02-24       Impact factor: 3.390

  5 in total

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