Human Mutation - 2015

200 articles | Last updated: 2025-12-03 14:12:57
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T A T A T A T A
Issue Information
The Genome Editing Revolution in Livestock Marches on
Cover Image, Volume 37, Issue 1
Genetic Variants in MicroRNAs and Their Binding Sites Are Associated with the Risk of Parkinson Disease
Multiallelic Positions in the Human Genome: Challenges for Genetic Analyses
<i>MCM3AP</i>and<i>POMP</i>Mutations Cause a DNA-Repair and DNA-Damage-Signaling Defect in an Immunodeficient Child
<i>HMOX2</i>Functions as a Modifier Gene for High-Altitude Adaptation in Tibetans
Identification and Characterization of a Novel Constitutional<i>PIK3CA</i>Mutation in a Child Lacking the Typical Segmental Overgrowth of “<i>PIK3CA</i>-Related Overgrowth Spectrum”
PEX6 is Expressed in Photoreceptor Cilia and Mutated in Deafblindness with Enamel Dysplasia and Microcephaly
Long-Read Single Molecule Real-Time Full Gene Sequencing of Cytochrome P450-2D6
A Newly Identified Missense Mutation in<i>FARS2</i>Causes Autosomal-Recessive Spastic Paraplegia
Serial Hunt for Ciliary Genes in Complex Syndromes
Correction of a Cystic Fibrosis Splicing Mutation by Antisense Oligonucleotides
Mechanisms for the Generation of Two Quadruplications Associated with Split-Hand Malformation
A Zebrafish Loss-of-Function Model for Human CFAP53 Mutations Reveals Its Specific Role in Laterality Organ Function
Clinical Sensitivity of Cystic Fibrosis Mutation Panels in a Diverse Population
TMEM107 Is a Critical Regulator of Ciliary Protein Composition and Is Mutated in Orofaciodigital Syndrome
Whole<i>USH2A</i>Gene Sequencing Identifies Several New Deep Intronic Mutations
Mandibulofacial Dysostosis with Microcephaly: Mutation and Database Update
SMPD1 Mutation Update: Database and Comprehensive Analysis of Published and Novel Variants
Alternative Splicing in the Human<i>PMP22</i>Gene: Implications in CMT1A Neuropathy
From Whole Gene Deletion to Point Mutations of<i>EP300</i>-Positive Rubinstein-Taybi Patients: New Insights into the Mutational Spectrum and Peculiar Clinical Hallmarks
A Role for Non-B DNA Forming Sequences in Mediating Microlesions Causing Human Inherited Disease
A Standardized DNA Variant Scoring System for Pathogenicity Assessments in Mendelian Disorders
Identification and Functional Characterization of<i>CLCN1</i>Mutations Found in Nondystrophic Myotonia Patients
Diagnostic Exome Sequencing Identifies a Novel Gene,<i>EMILIN1</i>, Associated with Autosomal‐Dominant Hereditary Connective Tissue Disease
Mutation Update of<i>ARSA</i>and<i>PSAP</i>Genes Causing Metachromatic Leukodystrophy
Neurofibromatosis Type 1 Without Neurofibromas: Genotype-Phenotype Correlations in NF1
Purifying Selection of mtDNA and the Shape of Human Evolution
Contents
Efficient Generation of Gene-Modified Pigs Harboring Precise Orthologous Human Mutation via CRISPR/Cas9-Induced Homology-Directed Repair in Zygotes
wKinMut-2: Identification and Interpretation of Pathogenic Variants in Human Protein Kinases
Assessing the Pathogenicity of Insertion and Deletion Variants with the Variant Effect Scoring Tool (VEST‐Indel)
Targeted Droplet-Digital PCR as a Tool for Novel Deletion Discovery at the DFNB1 Locus
Mutation Update for<i>COL2A1</i>Gene Variants Associated with Type II Collagenopathies
Functional and Clinical Consequences of Novel α-Galactosidase A Mutations in Fabry Disease
Putative Prostate Cancer Risk SNP in an Androgen Receptor‐Binding Site of the Melanophilin Gene Illustrates Enrichment of Risk SNPs in Androgen Receptor Target Sites
Rare Variants in<i>PLD3</i>Do Not Affect Risk for Early-Onset Alzheimer Disease in a European Consortium Cohort European Consortium Cohort
Variants in<i>TRIM44</i>Cause Aniridia by Impairing<i>PAX6</i>Expression
Network-Informed Gene Ranking Tackles Genetic Heterogeneity in Exome-Sequencing Studies of Monogenic Disease
Simultaneous Whole Mitochondrial Genome Sequencing with Short Overlapping Amplicons Suitable for Degraded DNA Using the Ion Torrent Personal Genome Machine
Pulmonary Arterial Hypertension: A Current Perspective on Established and Emerging Molecular Genetic Defects
Performance of In Silico Tools for the Evaluation of<i>UGT1A1</i>Missense Variants
Contents
Mitigating False-Positive Associations in Rare Disease Gene Discovery
Targeted Next‐Generation Sequencing Analysis of 1,000 Individuals with Intellectual Disability
Functional Analysis of BARD1 Missense Variants in Homology-Directed Repair of DNA Double Strand Breaks
Classification of Amino Acid Substitutions in Mismatch Repair Proteins Using PON-MMR2
Hidden Genetic Variation in LCA9‐Associated Congenital Blindness Explained by 5′UTR Mutations and Copy‐Number Variations of <i>NMNAT1</i>
Defects in tRNA Anticodon Loop 2′-<i>O</i>-Methylation Are Implicated in Nonsyndromic X-Linked Intellectual Disability due to Mutations in<i>FTSJ1</i>
<i>GMPPB</i>-Associated Dystroglycanopathy: Emerging Common Variants with Phenotype Correlation
Targeted Resequencing of 29 Candidate Genes and Mouse Expression Studies Implicate<i>ZIC3</i>and<i>FOXF1</i>in Human VATER/VACTERL Association
A Novel Mutation in<i>RPL10</i>(Ribosomal Protein L10) Causes X-Linked Intellectual Disability, Cerebellar Hypoplasia, and Spondylo-Epiphyseal Dysplasia
Beckwith-Wiedemann Syndrome Revisited
Contents
Use of Model Organism and Disease Databases to Support Matchmaking for Human Disease Gene Discovery
The Matchmaker Exchange: A Platform for Rare Disease Gene Discovery
Expanding the Molecular and Clinical Phenotype of SSR4-CDG
The Matchmaker Exchange API: Automating Patient Matching Through the Exchange of Structured Phenotypic and Genotypic Profiles
<i>SMAD2</i>Mutations Are Associated with Arterial Aneurysms and Dissections
<i>DOCK6</i>Mutations Are Responsible for a Distinct Autosomal-Recessive Variant of Adams-Oliver Syndrome Associated with Brain and Eye Anomalies
Fine Time Scaling of Purifying Selection on Human Nonsynonymous mtDNA Mutations Based on the Worldwide Population Tree and Mother-Child Pairs
A New Homozygous<i>IGF1R</i>Variant Defines a Clinically Recognizable Incomplete Dominant form of SHORT Syndrome
PhenomeCentral: A Portal for Phenotypic and Genotypic Matchmaking of Patients with Rare Genetic Diseases
Participant-Driven Matchmaking in the Genomic Era
Novel<i>COCH</i>p.V123E Mutation, Causative of DFNA9 Sensorineural Hearing Loss and Vestibular Disorder, Shows Impaired Cochlin Post-Translational Cleavage and Secretion
The Genomic Birthday Paradox: How Much Is Enough?
Cafe Variome: General-Purpose Software for Making Genotype-Phenotype Data Discoverable in Restricted or Open Access Contexts
Facilitating Collaboration in Rare Genetic Disorders Through Effective Matchmaking in DECIPHER
Matching Two Independent Cohorts Validates<i>DPH1</i>as a Gene Responsible for Autosomal Recessive Intellectual Disability with Short Stature, Craniofacial, and Ectodermal Anomalies
Data Sharing in the Undiagnosed Diseases Network
Novel Homozygous Mutation of the Internal Translation Initiation Start Site of<i>VHL</i>is Exclusively Associated with Erythrocytosis: Indications for Distinct Functional Roles of von Hippel-Lindau Tu
GeneMatcher: A Matching Tool for Connecting Investigators with an Interest in the Same Gene
Concurrent DNA Copy-Number Alterations and Mutations in Genes Related to Maintenance of Genome Stability in Uninvolved Mammary Glandular Tissue from Breast Cancer Patients
Contents
Novel<i>COL2A1</i>Variant (c.619G&gt;A, p.Gly207Arg) Manifesting as a Phenotype Similar to Progressive Pseudorheumatoid Dysplasia and Spondyloepiphyseal Dysplasia, Stanescu Type
GenomeConnect: Matchmaking Between Patients, Clinical Laboratories, and Researchers to Improve Genomic Knowledge
Complex Multiple-Nucleotide Substitution Mutations Causing Human Inherited Disease Reveal Novel Insights into the Action of Translesion Synthesis DNA Polymerases
High Incidence of Noonan Syndrome Features Including Short Stature and Pulmonic Stenosis in Patients carrying NF1 Missense Mutations Affecting p.Arg1809: Genotype–Phenotype Correlation
Activating Mutations Affecting the Dbl Homology Domain of SOS2 Cause Noonan Syndrome
Innovative Genomic Collaboration Using the GENESIS (GEM.app) Platform
GeneMatcher Aids in the Identification of a New Malformation Syndrome with Intellectual Disability, Unique Facial Dysmorphisms, and Skeletal and Connective Tissue Abnormalities Caused by De Novo Varia
<i>POLD1</i>Germline Mutations in Patients Initially Diagnosed with Werner Syndrome
Mediterranean Founder Mutation Database (MFMD): Taking Advantage from Founder Mutations in Genetics Diagnosis, Genetic Diversity and Migration History of the Mediterranean Population
In Memoriam: Richard G.H. Cotton (1940-2015)
<i>FAS</i>Gene Copy Numbers are Associated with Susceptibility to Behçet Disease and VKH Syndrome in Han Chinese Han Chinese
Rare Variants in the Epithelial Cadherin Gene Underlying the Genetic Etiology of Nonsyndromic Cleft Lip with or without Cleft Palate
<i>WDR73</i>Mutations Cause Infantile Neurodegeneration and Variable Glomerular Kidney Disease
Evaluation of Hybridization Capture Versus Amplicon‐Based Methods for Whole‐Exome Sequencing
Amplicon Resequencing Identified Parental Mosaicism for Approximately 10% of “ <i>de novo</i> ” <i>SCN1A</i> Mutations in Children with Dravet Syndrome
15q Maternal Duplication and Neurodevelopmental Disorders;<i>UBE3A</i>is the Key
Contents
<i>KIAA0586</i>is Mutated in Joubert Syndrome
Choline Acetyltransferase Mutations Causing Congenital Myasthenic Syndrome: Molecular Findings and Genotype-Phenotype Correlations
Mutations of the Imprinted<i>CDKN1C</i>Gene as a Cause of the Overgrowth Beckwith-Wiedemann Syndrome: Clinical Spectrum and Functional Characterization
Nonsyndromic Early-Onset Cone-Rod Dystrophy and Limb-Girdle Muscular Dystrophy in a Consanguineous Israeli Family are Caused by Two Independent yet Linked Mutations in<i>ALMS1</i>and<i>DYSF</i>
Somatic Mutations in Catalytic Core of<i>POLK</i>Reported in Prostate Cancer Alter Translesion DNA Synthesis
Rapid Detection of Rare Deleterious Variants by Next Generation Sequencing with Optional Microarray SNP Genotype Data
Microdeletions of<i>ELP4</i>Are Associated with Language Impairment, Autism Spectrum Disorder, and Mental Retardation
The Effects of PMM2-CDG-Causing Mutations on the Folding, Activity, and Stability of the PMM2 Protein
Disclosing the Hidden Structure and Underlying Mutational Mechanism of a Novel Type of Duplication CNV Responsible for Hereditary Multiple Osteochondromas
Contents
Getting It Right with Lynch Syndrome Genetic and Phenotypic Diagnosis
Loss of Function Variants in Human<i>PNPLA8</i>Encoding Calcium-Independent Phospholipase A<sub>2</sub>γ Recapitulate the Mitochondriopathy of the Homologous Null Mouse
A Mitochondrial Translation Defect Identified by Whole-Exome Sequencing Expands the Phenotypic Spectrum for<i>MARS2</i>
Whole-Genome Sequencing and Integrative Genomic Analysis Approach on Two 22q11.2 Deletion Syndrome Family Trios for Genotype to Phenotype Correlations
Comparison of Exome and Genome Sequencing Technologies for the Complete Capture of Protein‐Coding Regions
Molecular Outcome, Prediction, and Clinical Consequences of Splice Variants in<i>COL1A1</i>, Which Encodes the proα1(I) Chains of Type I Procollagen
The DNA Bank: High-Security Bank Accounts to Protect and Share Your Genetic Identity
Molecular Diversity and Associated Phenotypic Spectrum of Germline<i>CBL</i>Mutations
Structural and Functional Impact of Parkinson Disease-Associated Mutations in the E3 Ubiquitin Ligase Parkin
A Novel<i>KCNJ13</i>Nonsense Mutation and Loss of Kir7.1 Channel Function Causes Leber Congenital Amaurosis (LCA16)
McArdle Disease: Update of Reported Mutations and Polymorphisms in the<i>PYGM</i>Gene
Further Confirmation of Germline Glioma Risk Variant rs78378222 in<i>TP53</i>and Its Implication in Tumor Tissues via Integrative Analysis of TCGA Data
Performant Mutation Identification Using Targeted Next-Generation Sequencing of 14 Thoracic Aortic Aneurysm Genes
The KM-parkin-DB: A Sub-set<i>MutationView</i>Database Specialized for<i>PARK2</i>(PARKIN) Variants
Mutation Update of the<i>CLCN5</i>Gene Responsible for Dent Disease 1
An Interdomain<i>KCNH2</i>Mutation Produces an Intermediate Long QT Syndrome
Skipping Skin Disease
The Importance of Proper Testing of Predictor Performance
Contents
15q11.2 Duplication Encompassing Only the<i>UBE3A</i>Gene Is Associated with Developmental Delay and Neuropsychiatric Phenotypes
Evaluation of CADD Scores in Curated Mismatch Repair Gene Variants Yields a Model for Clinical Validation and Prioritization
A Gain-of-Function Mutation in<i>NALCN</i>in a Child with Intellectual Disability, Ataxia, and Arthrogryposis
<i>DOCK6</i>Mutations Are Responsible for a Distinct Autosomal-Recessive Variant of Adams-Oliver Syndrome Associated with Brain and Eye Anomalies
Alström Syndrome: Mutation Spectrum of<i>ALMS1</i>
The Ultimate Guide to DMD Variants
Shaken, Not Stirred Collagen in Corneal Dystrophy
Contents
Hemizygosity for<i>SMCHD1</i>in Facioscapulohumeral Muscular Dystrophy Type 2: Consequences for 18p Deletion Syndrome
Functional Complementation Assay for 47 <i>MUTYH</i> Variants in a <i>MutY</i> ‐Disrupted <i>Escherichia Coli</i> Strain
<i>LMNA</i>Mutation c.917T&gt;G (p.L306R) Leads to Deleterious Hyper-Assembly of Lamin A/C and Associates with Severe Right Ventricular Cardiomyopathy and Premature Aging
Characterization of All Possible Single-Nucleotide Change Caused Amino Acid Substitutions in the Kinase Domain of Bruton Tyrosine Kinase
A New Spin on Digenic Inheritance: Retrotransposition in Rotor Syndrome
Towards Replacement of Sanger Sequencing with Next-Generation Sequencing in the Clinical Laboratory
Contents
Response to: Does IARS2 Deficiency Cause an Intrinsic Disorder of Bone Development (Skeletal Dysplasia) or Are the Reported Skeletal Changes Secondary to Growth Hormone Deficiency and Neuromuscular In
Does IARS2 Deficiency Cause an Intrinsic Disorder of Bone Development (Skeletal Dysplasia) or Are the Reported Skeletal Changes Secondary to Growth Hormone Deficiency and Neuromuscular Involvement?
Next-Generation Diagnostics: Gene Panel, Exome, or Whole Genome?
Understanding the Implications of Mitochondrial DNA Variation in the Health of Black Southern African Populations: The 2014 Workshop
Lynch Syndrome Associated with Two <i>MLH1</i> Promoter Variants and Allelic Imbalance of <i>MLH1</i> Expression
Heterozygous Deletion of<i>FOXA2</i>Segregates with Disease in a Family with Heterotaxy, Panhypopituitarism, and Biliary Atresia
amamutdb.no: A Relational Database for<i>MAN2B1</i>Allelic Variants that Compiles Genotypes, Clinical Phenotypes, and Biochemical and Structural Data of Mutant MAN2B1 in α-Mannosidosis
The<i>SCN1A</i>Mutation Database: Updating Information and Analysis of the Relationships among Genotype, Functional Alteration, and Phenotype
Loss-of-Function<i>FANCL</i>Mutations Associate with Severe Fanconi Anemia Overlapping the VACTERL Association
Novel, Compound Heterozygous, Single-Nucleotide Variants in<i>MARS2</i>Associated with Developmental Delay, Poor Growth, and Sensorineural Hearing Loss
Insights into Severe 5,10-Methylenetetrahydrofolate Reductase Deficiency: Molecular Genetic and Enzymatic Characterization of 76 Patients
Expanding the Mutation Spectrum Affecting αIIbβ3 Integrin in Glanzmann Thrombasthenia: Screening of the<i>ITGA2B</i>and<i>ITGB3</i>Genes in a Large International Cohort
Update and Mutational Analysis of<i>SLC20A2</i>: A Major Cause of Primary Familial Brain Calcification
The Transcription-Coupled Repair Protein ERCC6/CSB Also Protects Against Repeat Expansion in a Mouse Model of the Fragile X Premutation
GeneYenta: A Phenotype­Based Rare Disease Case Matching Tool Based on Online Dating Algorithms for the Acceleration of Exome Interpretation
Oncotator: Cancer Variant Annotation Tool
Genetic Heterogeneity and Clinical Variability in Musculocontractural Ehlers-Danlos Syndrome Caused by Impaired Dermatan Sulfate Biosynthesis
Differential Dimerization of Variants Linked to Enhanced S-Cone Sensitivity Syndrome (ESCS) Located in the NR2E3 Ligand-Binding Domain
Heterozygous Mutations in Natriuretic Peptide Receptor-B (<i>NPR2</i>) Gene as a Cause of Short Stature
The Evaluation of Tools Used to Predict the Impact of Missense Variants Is Hindered by Two Types of Circularity
New Tools for Mendelian Disease Gene Identification: PhenoDB Variant Analysis Module; and GeneMatcher, a Web-Based Tool for Linking Investigators with an Interest in the Same Gene
mit-o-matic: A Comprehensive Computational Pipeline for Clinical Evaluation of Mitochondrial Variations from Next-Generation Sequencing Datasets
Mutations in Collagen, Type XVII, Alpha 1 (<i>COL17A1</i>) Cause Epithelial Recurrent Erosion Dystrophy (ERED)
The ARVD/C Genetic Variants Database: 2014 Update
wKGGSeq: A Comprehensive Strategy-Based and Disease-Targeted Online Framework to Facilitate Exome Sequencing Studies of Inherited Disorders
On Human Disease-Causing Amino Acid Variants: Statistical Study of Sequence and Structural Patterns
Exon-Specific U1s Correct SPINK<i>5</i>Exon 11 Skipping Caused by a Synonymous Substitution that Affects a Bifunctional Splicing Regulatory Element
AmpliVar: Mutation Detection in High-Throughput Sequence from Amplicon-Based Libraries
<i>De Novo</i>Heterozygous Mutations in<i>SMC3</i>Cause a Range of Cornelia de Lange Syndrome-Overlapping Phenotypes
Contents
Navigating through Genomics Data to Deliver Testable Predictions
Hidden Mutations in Cornelia de Lange Syndrome Limitations of Sanger Sequencing in Molecular Diagnostics
Shotgun Approach to Functional Annotation of Genes
Mutation in The Nuclear-Encoded Mitochondrial Isoleucyl-tRNA Synthetase<i>IARS2</i>in Patients with Cataracts, Growth Hormone Deficiency with Short Stature, Partial Sensorineural Deafness, and Periphe
Mutations in<i>TAX1BP3</i>Cause Dilated Cardiomyopathy with Septo-Optic Dysplasia
Identification of Variants in the 4q35 Gene<i>FAT1</i>in Patients with a Facioscapulohumeral Dystrophy-Like Phenotype
The TREAT-NMD DMD Global Database: Analysis of More than 7,000 Duchenne Muscular Dystrophy Mutations
Germline &amp; Somatic Mosaicism: The 2014 Annual Scientific Meeting of the Human Genome Variation Society
Heterozygous Triplication of Upstream Regulatory Sequences Leads to Dysregulation of Matrix Metalloproteinase 19 in Patients with Cavitary Optic Disc Anomaly
Recessive Inheritance of Population-Specific Intronic LINE-1 Insertion Causes a Rotor Syndrome Phenotype
Gene Conversion Between Cationic Trypsinogen (<i>PRSS1</i> ) and the Pseudogene Trypsinogen 6 (<i>PRSS3P2</i> ) in Patients with Chronic Pancreatitis
Can We Afford to Sequence Every Newborn Baby's Genome?
Rare <i>LRP6</i> Variants Identified in Spina Bifida Patients
Optimizing the Molecular Diagnosis of GALNS: Novel Methods to Define and Characterize Morquio-A Syndrome-Associated Mutations
Concurrent Nucleotide Substitution Mutations in the Human Genome Are Characterized by a Significantly Decreased Transition/Transversion Ratio
Somatic MMR Gene Mutations as a Cause for MSI-H Sebaceous Neoplasms in Muir-Torre Syndrome-Like Patients
Functional Analysis of<i>FOXE3</i>Mutations Causing Dominant and Recessive Ocular Anterior Segment Disease
Benefits and Burdens of Using a SNP Array in Pregnancies at Increased Risk for the Common Aneuploidies
Loss of Function Variants in Human <i>PNPLA8</i> Encoding Calcium-Independent Phospholipase A<sub>2</sub> γ Recapitulate the Mitochondriopathy of the Homologous Null Mouse
Novel Mutations in the DYNC1H1 Tail Domain Refine the Genetic and Clinical Spectrum of Dyneinopathies
Flexible, Scalable, and Efficient Targeted Resequencing on a Benchtop Sequencer for Variant Detection in Clinical Practice
Inhibition of RAS Activation Due to a Homozygous Ezrin Variant in Patients with Profound Intellectual Disability
Mutations in <i>CCDC11</i> , which Encodes a Coiled-Coil Containing Ciliary Protein, Causes <i>Situs Inversus</i> Due to Dysmotility of Monocilia in the Left-Right Organizer
Genome Annotation by Shotgun Inactivation of a Native Gene in Hemizygous Cells: Application to<i>BRCA2</i>with Implication of Hypomorphic Variants
Single Nucleotide Differences (SNDs) Continue to Contaminate the dbSNP Database With Consequences for Human Genomics and Health
High-Resolution Breakpoint Analysis Provides Evidence for the Sequence-Directed Nature of Genome Rearrangements in Hereditary Disorders
Identification and<i>In Vivo</i>Functional Characterization of Novel Compound Heterozygous<i>BMP1</i>Variants in Osteogenesis Imperfecta
Identification and Functional Analysis of a SLC33A1: c.339T&gt;G (p.Ser113Arg) Variant in the Original SPG42 Family
ECHS1 Mutations Cause Combined Respiratory Chain Deficiency Resulting in Leigh Syndrome
Two Siblings with Homozygous Pathogenic Splice-Site Variant in Mitochondrial Asparaginyl-tRNA Synthetase (<i>NARS2</i>)
VariSNP, A Benchmark Database for Variations From dbSNP
Functional Classification of <i>BRCA2</i> DNA Variants by Splicing Assays in a Large Minigene with 9 Exons
ProKinO: A Unified Resource for Mining the Cancer Kinome
Vanno: A Visualization-Aided Variant Annotation Tool