Literature DB >> 28692176

Phenotypic spectrum associated with de novo mutations in QRICH1 gene.

A Ververi1, M Splitt2, J C S Dean3, A F Brady1.   

Abstract

Rare de novo mutations represent a significant cause of idiopathic developmental delay (DD). The use of next-generation sequencing (NGS) has boosted the identification of de novo mutations in an increasing number of novel genes. Here we present 3 unrelated children with de novo loss-of-function (LoF) mutations in QRICH1, diagnosed through trio-based exome sequencing. QRICH1 encodes the glutamine-rich protein 1, which contains 1 caspase activation recruitment domain and is likely to be involved in apoptosis and inflammation. All 3 children had speech delay, learning difficulties, a prominent nose and a thin upper lip. In addition, 2 of them had mildly raised creatine kinase (CK) and 1 of them had autism. Despite their small number, the patients had a relatively consistent pattern of clinical features suggesting the presence of a QRICH1-associated phenotype. LoF mutations in QRICH1 are suggested as a novel cause of DD.
© 2017 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  zzm321990CK; zzm321990QRICH1; autism; developmental delay

Mesh:

Substances:

Year:  2017        PMID: 28692176     DOI: 10.1111/cge.13096

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  6 in total

1.  QRICH1 mutations cause a chondrodysplasia with developmental delay.

Authors:  Julian C Lui; Youn Hee Jee; Audrey Lee; Shanna Yue; Jacob Wagner; Deirdre E Donnelly; Karen S Vogt; Jeffrey Baron
Journal:  Clin Genet       Date:  2018-10-26       Impact factor: 4.438

2.  QRICH1 dictates the outcome of ER stress through transcriptional control of proteostasis.

Authors:  Kwontae You; Lingfei Wang; Chih-Hung Chou; Kai Liu; Toru Nakata; Alok Jaiswal; Junmei Yao; Ariel Lefkovith; Abdifatah Omar; Jacqueline G Perrigoue; Jennifer E Towne; Aviv Regev; Daniel B Graham; Ramnik J Xavier
Journal:  Science       Date:  2021-01-01       Impact factor: 47.728

3.  Novel pathogenic variants and multiple molecular diagnoses in neurodevelopmental disorders.

Authors:  Joanne Trinh; Krishna Kumar Kandaswamy; Martin Werber; Maximilian E R Weiss; Gabriela Oprea; Shivendra Kishore; Katja Lohmann; Arndt Rolfs
Journal:  J Neurodev Disord       Date:  2019-06-25       Impact factor: 4.025

Review 4.  The Pancreatic ß-cell Response to Secretory Demands and Adaption to Stress.

Authors:  Michael A Kalwat; Donalyn Scheuner; Karina Rodrigues-Dos-Santos; Decio L Eizirik; Melanie H Cobb
Journal:  Endocrinology       Date:  2021-11-01       Impact factor: 4.736

5.  Loss-of-function mutations in QRICH2 cause male infertility with multiple morphological abnormalities of the sperm flagella.

Authors:  Ying Shen; Feng Zhang; Fuping Li; Xiaohui Jiang; Yihong Yang; Xiaoliang Li; Weiyu Li; Xiang Wang; Juan Cheng; Mohan Liu; Xueguang Zhang; Guiping Yuan; Xue Pei; Kailai Cai; Fengyun Hu; Jianfeng Sun; Lanzhen Yan; Li Tang; Chuan Jiang; Wenling Tu; Jinyan Xu; Haojuan Wu; Weiqi Kong; Shuying Li; Ke Wang; Kai Sheng; Xudong Zhao; Huanxun Yue; Xiaoyu Yang; Wenming Xu
Journal:  Nat Commun       Date:  2019-01-25       Impact factor: 14.919

6.  Missed diagnoses: Clinically relevant lessons learned through medical mysteries solved by the Undiagnosed Diseases Network.

Authors:  Heidi Cope; Rebecca Spillmann; Jill A Rosenfeld; Elly Brokamp; Rebecca Signer; Kelly Schoch; Emily G Kelley; Jennifer A Sullivan; Ellen Macnamara; Sharyn Lincoln; Katie Golden-Grant; James P Orengo; Gary Clark; Lindsay C Burrage; Jennifer E Posey; Jaya Punetha; Amy Robertson; Joy Cogan; John A Phillips; Julian Martinez-Agosto; Vandana Shashi
Journal:  Mol Genet Genomic Med       Date:  2020-07-30       Impact factor: 2.183

  6 in total

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