Human Genetics - 2022

104 articles | Last updated: 2025-12-03 14:12:56
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Whole exome sequencing improves genetic diagnosis of fetal clubfoot
Mendelian randomization analysis reveals causal relationships between gut microbiome and optic neuritis
Quantitative assessment of low-level parental mosaicism of SNVs and CNVs in Waardenburg syndrome
KIF26A is mutated in the syndrome of congenital hydrocephalus with megacolon
CLEC16A interacts with retromer and TRIM27, and its loss impairs endosomal trafficking and neurodevelopment
Mate-pair genome sequencing reveals structural variants for idiopathic male infertility
Targeting de novo loss-of-function variants in constrained disease genes improves diagnostic rates in the 100,000 Genomes Project
Integrated exome and transcriptome analysis prioritizes MAP4K4 de novo frameshift variants in autism spectrum disorder as a novel disease–gene association
From collected stamps to hair locks: ethical and legal implications of testing DNA found on privately owned family artifacts
A homozygous AP3D1 missense variant in patients with sensorineural hearing loss as the leading manifestation
Investigating the shared genetic architecture and causal relationship between pain and neuropsychiatric disorders
The genomic analysis of current-day North African populations reveals the existence of trans-Saharan migrations with different origins and dates
The genetics of monogenic intestinal epithelial disorders
Hearing of Otof-deficient mice restored by trans-splicing of N- and C-terminal otoferlin
Overlapping pathogenic de novo CNVs in neurodevelopmental disorders and congenital anomalies impacting constraint genes regulating early development
SCIP: software for efficient clinical interpretation of copy number variants detected by whole-genome sequencing
Re: “Next generation sequencing in neonatology: what does it mean for the next generation?”
Genome-wide analyses of early-onset acute myocardial infarction identify 29 novel loci by whole genome sequencing
Identifying shared genetic factors underlying epilepsy and congenital heart disease in Europeans in Europeans
Variations in mitochondrial DNA coding and d-loop region are associated with early embryonic development defects in infertile women
Profiling human pathogenic repeat expansion regions by synergistic and multi-level impacts on molecular connections
Genome screening, reporting, and genetic counseling for healthy populations
The matrilineal ancestry of Nepali populations
Common genetic risk factors in ASD and ADHD co-occurring families
Large-scale pedigree analysis highlights rapidly mutating Y-chromosomal short tandem repeats for differentiating patrilineal relatives and predicting their degrees of consanguinity
PMEL is mutated in oculocutaneous albinism
Publisher Correction: Predicting functional consequences of mutations using molecular interaction network features
Publisher Correction: Revealing modifier variations characterizations for elucidating the genetic basis of human phenotypic variations
Retrotransposon insertion as a novel mutational cause of spinal muscular atrophy
Computational interpretation of human genetic variation
Publisher Correction: Gene expression levels modulate germline mutation rates through the compound effects of transcription-coupled repair and damage
Truncation mutations in MYRF underlie primary angle closure glaucoma
Dissection of mendelian predisposition and complex genetic architecture of craniovertebral junction malformation
Nonsense mutation in the novel PERCC1 gene as a genetic cause of congenital diarrhea and enteropathy Irish
Heterozygous variants in the DVL2 interaction region of DACT1 cause CAKUT and features of Townes–Brocks syndrome 2
RNA-seq analysis, targeted long-read sequencing and in silico prediction to unravel pathogenic intronic events and complicated splicing abnormalities in dystrophinopathy
Comprehensive analysis of microsatellite polymorphisms in human populations
Comprehensive interpretation of single-nucleotide substitutions in GJB2 reveals the genetic and phenotypic landscape of GJB2-related hearing loss
De novo mutations in the BMP signaling pathway in lambdoid craniosynostosis
Hemizygosity can reveal variant pathogenicity on the X-chromosome
Locus-level antagonistic selection shaped the polygenic architecture of human complex diseases
Contribution of whole genome sequencing in the molecular diagnosis of mosaic partial deletion of the NF1 gene in neurofibromatosis type 1
Special issue: Artificial intelligence in genomics
Clinical implications of genetic testing in familial intermediate and late-onset colorectal cancer
The heritability of vocal tract structures estimated from structural MRI in a large cohort of Dutch twins Dutch
Monogenic inflammatory bowel disease-genetic variants, functional mechanisms and personalised medicine in clinical practice
Clinical characteristics and comorbidities of COVID-19 in unvaccinated patients with Down syndrome: first year report in Brazil
Inherited metabolic disorders beyond the new generation sequencing era: the need for in-depth cellular and molecular phenotyping
A comprehensive genomic reporting structure for communicating all clinically significant primary and secondary findings
46,XY disorders of sex development: the use of NGS for prevalent variants
GATA4 and estrogen receptor alpha bind at SNPs rs9921222 and rs10794639 to regulate AXIN1 expression in osteoblasts
From first report to clinical trials: a bibliometric overview and visualization of the development of Angelman syndrome research
Fanconi anemia: current insights regarding epidemiology, cancer, and DNA repair
Homozygous missense mutation in CCDC155 disrupts the transmembrane distribution of CCDC155 and SUN1, resulting in non-obstructive azoospermia and premature ovarian insufficiency in humans
Editorial to special issue on Ethics in Genetics
Transmission ratio distortion of mutations in the master regulator of centriole biogenesis PLK4
Monogenic causes of pigmentary mosaicism
The clinical utility of polygenic risk scores in genomic medicine practices: a systematic review
Genetic genealogy uncovers a founder deletion mutation in the cerebral cavernous malformations 2 gene
Genome interpretation using in silico predictors of variant impact
Pharmacogenomics: the low-hanging fruit in the personalized medicine tree
Validating the knowledge bank approach for personalized prediction of survival in acute myeloid leukemia: a reproducibility study
Genomics and justice: mitigating the potential harms and inequities that arise from the implementation of genomics in medicine
Editorial to the Special Issue on “The molecular genetics of hearing and deafness”
Predicting genes from phenotypes using human phenotype ontology (HPO) terms
Next generation sequencing in neonatology: what does it mean for the next generation?
Predicting embryonic aneuploidy rate in IVF patients using whole-exome sequencing
Genetic testing for pediatric hearing loss: no time to waste
The genetic and phenotypic landscapes of Usher syndrome: from disease mechanisms to a new classification
Usher syndrome IIIA: a review of the disorder and preclinical research advances in therapeutic approaches
CD55-deficiency in Jews of Bukharan descent is caused by the Cromer blood type Dr(a−) variant Jews of Bukharan descent
The hearing-impaired patient: what the future holds
Autosomal dominant non-syndromic hearing loss maps to DFNA33 (13q34) and co-segregates with splice and frameshift variants in ATP11A, a phospholipid flippase gene
Signatures of genetic variation in human microRNAs point to processes of positive selection and population-specific disease risks
Update on CD164 and LMX1A genes to strengthen their causative role in autosomal dominant hearing loss
Challenges in translational machine learning
Advancing discovery in hearing research via biologist-friendly access to multi-omic data
Uterine fibroid polygenic risk score (PRS) associates and predicts risk for uterine fibroid
Usher syndrome type IV: clinically and molecularly confirmed by novel ARSG variants
Lack of NKG2D in MAGT1-deficient patients is caused by hypoglycosylation
DVPred: a disease-specific prediction tool for variant pathogenicity classification for hearing loss
Epimutation in inherited metabolic disorders: the influence of aberrant transcription in adjacent genes
An effector index to predict target genes at GWAS loci
Evolutionary history of type II transmembrane serine proteases involved in viral priming
C18orf32 loss-of-function is associated with a neurodevelopmental disorder with hypotonia and contractures
Evaluating the relevance of sequence conservation in the prediction of pathogenic missense variants
Trypsinogen (PRSS1 and PRSS2) gene dosage correlates with pancreatitis risk across genetic and transgenic studies: a systematic review and re-analysis
BMP3 is a novel locus involved in the causality of ocular coloboma
Heat increases full-length SMN splicing: promise for splice-augmenting therapies for SMA
Inactivating NHLH2 variants cause idiopathic hypogonadotropic hypogonadism and obesity in humans
Analysis of histone variant constraint and tissue expression suggests five potential novel human disease genes: H2AFY2, H2AFZ, H2AFY, H2AFV, H1F0
Genetic etiology of non-syndromic hearing loss in Europe
Amelioration of a neurodevelopmental disorder by carbamazepine in a case having a gain-of-function GRIA3 variant
Correction to: Genetic etiology of non-syndromic hearing loss in Latin America
AudioGene: refining the natural history of KCNQ4, GSDME, WFS1, and COCH-associated hearing loss
Biochemical analysis of novel NAA10 variants suggests distinct pathogenic mechanisms involving impaired protein N-terminal acetylation
Biallelic DNAH9 mutations are identified in Chinese patients with defective left–right patterning and cilia-related complex congenital heart disease Chinese
Genetic etiology of hearing loss in Iran
Fine human genetic map based on UK10K data set
Variants in CDH23 cause a broad spectrum of hearing loss: from non-syndromic to syndromic hearing loss as well as from congenital to age-related hearing loss Japanese population; East Asian population
Case series of congenital pseudarthrosis of the tibia unfulfilling neurofibromatosis type 1 diagnosis: 21% with somatic NF1 haploinsufficiency in the periosteum
SLC10A7, an orphan member of the SLC10 family involved in congenital disorders of glycosylation
Interpretable generative deep learning: an illustration with single cell gene expression data
SNP characteristics and validation success in genome wide association studies