Literature DB >> 30496831

Intrafamilial variability of XYLT2-related spondyloocular syndrome.

Naz Guleray1, Pelin Ozlem Simsek Kiper2, Gulen Eda Utine2, Koray Boduroglu3, Mehmet Alikasifoglu3.   

Abstract

Spondyloocular syndrome is characterized by generalized osteoporosis, multiple fractures and severe ocular findings. The causative XYLT2 mutations have recently been identified with the use of whole exome sequencing. We report on two siblings with spondyloocular syndrome who presented with varying clinical severity. A novel XYLT2 missense mutation was detected in a region evolutionary conserved across the species. This report along with the previous reports demonstrates that variable expressivity may be possible even within the same family. These two siblings with a novel mutation further expand the clinical and mutational spectrum of spondyloocular syndrome.
Copyright © 2018 Elsevier Masson SAS. All rights reserved.

Entities:  

Keywords:  Cataract; Osteoporosis; Retinal detachment; Spondyloocular syndrome; XYLT2

Year:  2018        PMID: 30496831     DOI: 10.1016/j.ejmg.2018.11.019

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  5 in total

Review 1.  The clinical and mutational spectrum of B3GAT3 linkeropathy: two case reports and literature review.

Authors:  Marlies Colman; Tim Van Damme; Elisabeth Steichen-Gersdorf; Franco Laccone; Sheela Nampoothiri; Delfien Syx; Brecht Guillemyn; Sofie Symoens; Fransiska Malfait
Journal:  Orphanet J Rare Dis       Date:  2019-06-13       Impact factor: 4.123

Review 2.  Further Defining the Phenotypic Spectrum of B3GAT3 Mutations and Literature Review on Linkeropathy Syndromes.

Authors:  Marco Ritelli; Valeria Cinquina; Edoardo Giacopuzzi; Marina Venturini; Nicola Chiarelli; Marina Colombi
Journal:  Genes (Basel)       Date:  2019-08-21       Impact factor: 4.096

3.  Spondyloocular Syndrome: A Novel XYLT2 Variant with Description of the Neonatal Phenotype.

Authors:  Gabriella Doddato; Alessandra Fabbiani; Chiara Fallerini; Mirella Bruttini; Theodora Hadjistilianou; Martino Landi; Caterina Coradeschi; Salvatore Grosso; Barbara Tomasini; Maria Antonietta Mencarelli; Alessandra Renieri; Francesca Ariani
Journal:  Front Genet       Date:  2021-12-03       Impact factor: 4.599

Review 4.  Chondrodysplasias With Multiple Dislocations Caused by Defects in Glycosaminoglycan Synthesis.

Authors:  Johanne Dubail; Valérie Cormier-Daire
Journal:  Front Genet       Date:  2021-06-16       Impact factor: 4.599

5.  Xylosyltransferase 2 deficiency and organ homeostasis.

Authors:  Beatrix Ferencz; Eduard Condac; Nabin Poudel; Maria Cristina Munteanu; Pulavendran Sivasami; Biswa Choudhury; Nandita Natasha Naidu; Fuming Zhang; Melanie Breshears; Robert J Linhardt; Myron E Hinsdale
Journal:  Glycoconj J       Date:  2020-09-23       Impact factor: 3.009

  5 in total

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