Literature DB >> 26827111

Added value of next generation gene panel analysis for patients with elevated methylmalonic acid and no clinical diagnosis following functional studies of vitamin B12 metabolism.

Mihaela Pupavac1, Xia Tian2, Jordan Chu1, Guoli Wang2, Yanming Feng2, Stella Chen2, Remington Fenter2, Victor W Zhang3, Jing Wang3, David Watkins1, Lee-Jun Wong3, David S Rosenblatt4.   

Abstract

Next generation sequencing (NGS) based gene panel testing is increasingly available as a molecular diagnostic approach for inborn errors of metabolism. Over the past 40 years patients have been referred to the Vitamin B12 Clinical Research Laboratory at McGill University for diagnosis of inborn errors of cobalamin metabolism by functional studies in cultured fibroblasts. DNA samples from patients in which no diagnosis was made by these studies were tested by a NGS gene panel to determine whether any molecular diagnoses could be made. 131 DNA samples from patients with elevated methylmalonic acid and no diagnosis following functional studies of cobalamin metabolism were analyzed using the 24 gene extended cobalamin metabolism NGS based panel developed by Baylor Miraca Genetics Laboratories. Gene panel testing identified two or more variants in a single gene in 16/131 patients. Eight patients had pathogenic findings, one had a finding of uncertain significance, and seven had benign findings. Of the patients with pathogenic findings, five had mutations in ACSF3, two in SUCLG1 and one in TCN2. Thus, the NGS gene panel allowed for the presumptive diagnosis of 8 additional patients for which a diagnosis was not made by the functional assays.
Copyright © 2016 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  Cobalamin metabolism; Diagnostic test; Functional studies; Methylmalonic acid; Next generation sequencing gene panel; Vitamin B(12)

Mesh:

Substances:

Year:  2016        PMID: 26827111     DOI: 10.1016/j.ymgme.2016.01.008

Source DB:  PubMed          Journal:  Mol Genet Metab        ISSN: 1096-7192            Impact factor:   4.797


  7 in total

1.  Hydrocephalus in cblC type methylmalonic acidemia.

Authors:  Kaihui Zhang; Min Gao; Guangyu Wang; Yingying Shi; Xiaoying Li; Yvqiang Lv; Guangye Zhang; Zhongtao Gai; Yi Liu
Journal:  Metab Brain Dis       Date:  2018-12-19       Impact factor: 3.584

2.  Population Reference Values for Serum Methylmalonic Acid Concentrations and Its Relationship with Age, Sex, Race-Ethnicity, Supplement Use, Kidney Function and Serum Vitamin B12 in the Post-Folic Acid Fortification Period.

Authors:  Vijay Ganji; Mohammad R Kafai
Journal:  Nutrients       Date:  2018-01-12       Impact factor: 5.717

3.  Combined Malonic and Methylmalonic Aciduria Due to ACSF3 Variants Results in Benign Clinical Course in Three Chinese Patients.

Authors:  Ping Wang; Jianbo Shu; Chunyu Gu; Xiaoli Yu; Jie Zheng; Chunhua Zhang; Chunquan Cai
Journal:  Front Pediatr       Date:  2021-11-25       Impact factor: 3.418

Review 4.  Biomarkers and Algorithms for the Diagnosis of Vitamin B12 Deficiency.

Authors:  Luciana Hannibal; Vegard Lysne; Anne-Lise Bjørke-Monsen; Sidney Behringer; Sarah C Grünert; Ute Spiekerkoetter; Donald W Jacobsen; Henk J Blom
Journal:  Front Mol Biosci       Date:  2016-06-27

5.  Improving the diagnosis of cobalamin and related defects by genomic analysis, plus functional and structural assessment of novel variants.

Authors:  Sandra Brasil; Fátima Leal; Ana Vega; Rosa Navarrete; María Jesús Ecay; Lourdes R Desviat; Casandra Riera; Natàlia Padilla; Xavier de la Cruz; Mari Luz Couce; Elena Martin-Hernández; Ana Morais; Consuelo Pedrón; Luis Peña-Quintana; Miriam Rigoldi; Norma Specola; Isabel Tavares de Almeida; Inmaculada Vives; Raquel Yahyaoui; Pilar Rodríguez-Pombo; Magdalena Ugarte; Celia Pérez-Cerda; Begoña Merinero; Belén Pérez
Journal:  Orphanet J Rare Dis       Date:  2018-07-24       Impact factor: 4.123

6.  Brain metabolism and neurological symptoms in combined malonic and methylmalonic aciduria.

Authors:  Sara Tucci
Journal:  Orphanet J Rare Dis       Date:  2020-01-22       Impact factor: 4.123

7.  Identification of transcobalamin deficiency with two novel mutations in the TCN2 gene in a Chinese girl with abnormal immunity: a case report.

Authors:  Shihong Zhan; Fangfang Cheng; Hailong He; Shaoyan Hu; Xing Feng
Journal:  BMC Pediatr       Date:  2020-10-06       Impact factor: 2.125

  7 in total

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