Genetics in Medicine - 2019

445 articles | Last updated: 2025-12-03 14:12:56
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We must now put in place an updated, comprehensive newborn screening program for deaf and hard-of-hearing infants
News
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Correction: Nonrandom occurrence of multiple de novo coding variants in a proband indicates the existence of an oligogenic model in autism
Correction: Genomic knowledge in the context of diagnostic exome sequencing: changes over time, persistent subgroup differences, and associations with psychological sequencing outcomes
Oncogenic effects of germline variants in lysosomal storage disease genes
Addendum: A practice guideline from the American College of Medical Genetics and Genomics and the National Society of Genetic Counselors: referral indications for cancer predisposition assessment
Correction: The value of diagnostic testing for parents of children with rare genetic diseases
Response to Veenstra et al.
Cost-effectiveness of population genomic screening
Extrusion pump ABCC1 was first linked with nonsyndromic hearing loss in humans by stepwise genetic analysis
In This Issue
News
Correction: Variants in MED12L, encoding a subunit of the Mediator kinase module, are responsible for intellectual disability associated with transcriptional defect
Liver fibrosis during clinical ascertainment of glycogen storage disease type III: a need for improved and systematic monitoring
DYRK1A-related intellectual disability: a syndrome associated with congenital anomalies of the kidney and urinary tract
The Liberfarb syndrome, a multisystem disorder affecting eye, ear, bone, and brain development, is caused by a founder pathogenic variant in the PISD gene
Chronic kidney disease in propionic acidemia
ACMG response to Nussbaum et al. letter on ACMG policy statement: the use of secondary findings recommendations for general population screening: a policy statement of the ACMG
The value of diagnostic testing for parents of children with rare genetic diseases
CTCF variants in 39 individuals with a variable neurodevelopmental disorder broaden the mutational and clinical spectrum
Response to “The use of ACMG secondary findings recommendations for general population screening: a policy statement of the American College of Medical Genetics and Genomics (ACMG)”
Correction: Delivering genomic medicine in the UK National Health Service: a systematic review and narrative synthesis
Genomic databases, subpoenas, and Certificates of Confidentiality
A cost-effectiveness model of genetic testing and periodical clinical screening for the evaluation of families with dilated cardiomyopathy
ADDENDUM: Genetic counseling and testing for Alzheimer disease: joint practice guidelines of the American College of Medical Genetics and the National Society of Genetic Counselors
Correction: Inaccuracies and shortcomings in “Adherence of cell-free DNA noninvasive prenatal screens to ACMG recommendations”
Genetic data partnerships: academic publications with privately owned or generated genetic data
An alternative approach to establishing unbiased colorectal cancer risk estimation in Lynch syndrome
Correction: A proposal for comprehensive newborn hearing screening to improve identification of deaf and hard-of-hearing children
Maternal copy-number variations in the DMD gene as secondary findings in noninvasive prenatal screening
Psychological outcomes related to exome and genome sequencing result disclosure: a meta-analysis of seven Clinical Sequencing Exploratory Research (CSER) Consortium studies
Correction: SMAD6 is frequently mutated in nonsyndromic radioulnar synostosis
Responsibility, culpability, and parental views on genomic testing for seriously ill children
Delivering genomic medicine in the United Kingdom National Health Service: a systematic review and narrative synthesis
Meta-analysis and multidisciplinary consensus statement: exome sequencing is a first-tier clinical diagnostic test for individuals with neurodevelopmental disorders
Biparental inheritance of mitochondrial DNA in humans is not a common phenomenon
A proposal for comprehensive newborn hearing screening to improve identification of deaf and hard-of-hearing children
PEDIA: prioritization of exome data by image analysis
Inaccuracies and shortcomings in “Adherence of cell-free DNA noninvasive prenatal screens to ACMG recommendations”
Response to Johansen Taber et al.
Variants in MED12L, encoding a subunit of the mediator kinase module, are responsible for intellectual disability associated with transcriptional defect
Low-level mosaicism in tuberous sclerosis complex: prevalence, clinical features, and risk of disease transmission
News
Reporting of race in genome and exome sequencing studies of cancer: a scoping review of the literature
Consensus interpretation of the p.Met34Thr and p.Val37Ile variants in GJB2 by the ClinGen Hearing Loss Expert Panel
In This Issue
Improving recommendations for genomic medicine: building an evolutionary process from clinical practice advisory documents to guidelines
Developmental outcomes of children with Duarte galactosemia: exploring the bases of an apparent contradiction in the literature
Androgenetic chimerism as an etiology for Beckwith–Wiedemann syndrome: diagnosis and management
Considerations for clinical curation, classification, and reporting of low-penetrance and low effect size variants associated with disease risk
Technical laboratory standards for interpretation and reporting of acquired copy-number abnormalities and copy-neutral loss of heterozygosity in neoplastic disorders: a joint consensus recommendation
SMAD6 is frequently mutated in nonsyndromic radioulnar synostosis
Correction: ClinGen expert clinical validity curation of 164 hearing loss gene–disease pairs
Phenotypic distinctions between mosaic forms of tuberous sclerosis complex
Chromosomal microarray vs. NIPS: analysis of 5541 low-risk pregnancies
Estimating prevalence for limb-girdle muscular dystrophy based on public sequencing databases
Significant contribution of intragenic deletions to ARID1B mutation spectrum
Response to Gorokhova et al.
Genotype–phenotype associations among panel-based TP53+ subjects
Genomic education for the next generation of health-care providers
Long-term economic impacts of exome sequencing for suspected monogenic disorders: diagnosis, management, and reproductive outcomes
The proportion of endometrial cancers associated with Lynch syndrome: a systematic review of the literature and meta-analysis
Clinical characteristics and genotypes in the ADVANCE baseline data set, a comprehensive cohort of US children and adolescents with Pompe disease
Correction: Sequencing as a first-line methodology for cystic fibrosis carrier screening
Including the blind community in precision medicine research: findings from a national survey and recommendations
Biallelic variants in the transcription factor PAX7 are a new genetic cause of myopathy
International Porphyria Molecular Diagnostic Collaborative: an evidence-based database of verified pathogenic and benign variants for the porphyrias
Targeted gene sequencing in 6994 individuals with neurodevelopmental disorder with epilepsy
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News
Correction: Genetic evaluation of cardiomyopathy: a clinical practice resource of the American College of Medical Genetics and Genomics(ACMG)
Correction: Variant classification changes over time in BRCA1 and BRCA2
Correction: Evaluation of the cost and effectiveness of diverse recruitment methods for a genetic screening study
Response to Yang et al.
How should we correctly interpret biallelic germline truncating variant of MLH3 in hereditary colorectal cancer?
Variant recurrence in neurodevelopmental disorders: the use of publicly available genomic data identifies clinically relevant pathogenic missense variants
Sequencing as a first-line methodology for cystic fibrosis carrier screening
Recessive variants in ZNF142 cause a complex neurodevelopmental disorder with intellectual disability, speech impairment, seizures, and dystonia
ADDENDUM: Section E9 of the American College of Medical Genetics Technical Standards and Guidelines: Fluorescence in situ hybridization
Expert and lay perspectives on burden, risk, tolerability, and acceptability of clinical interventions for genetic disorders
Correction: Mitochondrial oxodicarboxylate carrier deficiency is associated with mitochondrial DNA depletion and spinal muscular atrophy–like disease
Intrathecal enzyme replacement for Hurler syndrome: biomarker association with neurocognitive outcomes
Comorbidity landscape of the Danish patient population affected by chromosome abnormalities
The use of ACMG secondary findings recommendations for general population screening: a policy statement of the American College of Medical Genetics and Genomics (ACMG)
Points to consider in the reevaluation and reanalysis of genomic test results: a statement of the American College of Medical Genetics and Genomics (ACMG)
Pancreatic cancer and melanoma related perceptions and behaviors following disclosure of CDKN2A variant status as a research result
Aberrant DNA methylation as a diagnostic biomarker of diabetic embryopathy
Research participants’ preferences for receiving genetic risk information: a discrete choice experiment
Mutant RAMP2 causes primary open-angle glaucoma via the CRLR-cAMP axis
Letter: Relearning the 3 R’s? Reinterpretation, recontact, and return of genetic variants
Response to Knoppers et al.
Commentary: Expanded carrier screening: how much is too much?
Variant classification changes over time in BRCA1 and BRCA2
CRISPR in the North American popular press
PIGT-CDG, a disorder of the glycosylphosphatidylinositol anchor: description of 13 novel patients and expansion of the clinical characteristics
Identifying occult maternal malignancies from 1.93 million pregnant women undergoing noninvasive prenatal screening tests
ALPK1 missense pathogenic variant in five families leads to ROSAH syndrome, an ocular multisystem autosomal dominant disorder
All-cause mortality and survival in adults with 22q11.2 deletion syndrome
Successful recruitment and retention of diverse participants in a genomics clinical trial: a good invitation to a great party
A high-resolution X chromosome copy-number variation map in fertile females and women with primary ovarian insufficiency
Correction: Clinical and genetic spectrum of children with congenital diarrhea and enteropathy in China
Correction: Population genomic screening of all young adults in a health-care system: a cost-effectiveness analysis
Adherence of cell-free DNA noninvasive prenatal screens to ACMG recommendations
Biallelic loss-of-function P4HTM gene variants cause hypotonia, hypoventilation, intellectual disability, dysautonomia, epilepsy, and eye abnormalities (HIDEA syndrome)
News
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Evaluation of the cost and effectiveness of diverse recruitment methods for a genetic screening study
Challenges and lessons learned from clinical pharmacogenetic implementation of multiple gene–drug pairs across ambulatory care settings
A case–control collapsing analysis identifies retinal dystrophy genes associated with ophthalmic disease in patients with no pathogenic ABCA4 variants
What do we do now?: Responding to claims of germline gene editing in humans
De novo and biallelic DEAF1 variants cause a phenotypic spectrum
Mobility in osteogenesis imperfecta: a multicenter North American study
The Manchester International Consensus Group recommendations for the management of gynecological cancers in Lynch syndrome
From diagnostic yield to clinical impact: a pilot study on the implementation of prenatal exome sequencing in routine care
Clinical characteristics of 248 patients with Krabbe disease: quantitative natural history modeling based on published cases
Disability inclusion in precision medicine research: a first national survey
Rethinking the “open future” argument against predictive genetic testing of children
Clinical and genetic spectrum of children with congenital diarrhea and enteropathy in China
Correction: Toward automation of germline variant curation in clinical cancer genetics
ClinGen expert clinical validity curation of 164 hearing loss gene–disease pairs
A retrospective review of multiple findings in diagnostic exome sequencing: halF.A.re distinct and halF.A.re overlapping diagnoses
Multi-site investigation of strategies for the clinical implementation of CYP2D6 genotyping to guide drug prescribing
Atlas-CNV: a validated approach to call single-exon CNVs in the eMERGESeq gene panel
Nationwide population genetic screening improves outcomes of newborn screening for hearing loss in China
Correction: Analysis of fetal DNA in maternal plasma with markers designed for forensic DNA mixture resolution
Carrier frequency estimation of Zellweger spectrum disorder using ExAC database and bioinformatics tools
Estimating yields of prenatal carrier screening and implications for design of expanded carrier screening panels
Current conditions in medical genetics practice
Strategies for Improving Access to Hereditary Cancer Testing: Recommendations from Stakeholders
Elucidation of the phenotypic spectrum and genetic landscape in primary and secondary microcephaly
The transformation of medical genetics by clinical genomics: hubris meets humility
Atypical COL3A1 variants (glutamic acid to lysine) cause vascular Ehlers–Danlos syndrome with a consistent phenotype of tissue fragility and skin hyperextensibility
In This Issue
News
Clinical utility of noninvasive prenatal screening for expanded chromosome disease syndromes
Response to Wang et al.
Response to Roberts et al. 2018: cohort ascertainment and methods of analysis impact the association between cancer and genetic predisposition - the tale of breast cancer risk and Lynch syndrome genes
A data-driven evaluation of the size and content of expanded carrier screening panels
Correction: Experiences and perspectives on the return of secondary findings among genetic epidemiologists
Correction: Genomic mosaicism in the pathogenesis and inheritance of a Rett syndrome cohort
Correction: The phenotypic spectrum of WWOX-related disorders: 20 additional cases of WOREE syndrome and review of the literature
Toward automation of germline variant curation in clinical cancer genetics
Correction: BOADICEA: a comprehensive breast cancer risk prediction model incorporating genetic and nongenetic risk factors
Population genomic screening of all young adults in a health-care system: a cost-effectiveness analysis
Correction: Estimating the burden and economic impact of pediatric genetic disease
Phenotype of CM-AVM2 caused by variants in EPHB4: how much overlap with hereditary hemorrhagic telangiectasia (HHT)?
Clinical impact and cost-effectiveness of a 176-condition expanded carrier screen
Variants in TCF20 in neurodevelopmental disability: description of 27 new patients and review of literature
Genetic diversity and pathogenic variants as possible predictors of severity in a French sample of nonsyndromic heritable thoracic aortic aneurysms and dissections (nshTAAD)
SERPINA1 Z allele is associated with cystic fibrosis liver disease
Biallelic pathogenic variants in the lanosterol synthase gene LSS involved in the cholesterol biosynthesis cause alopecia with intellectual disability, a rare recessive neuroectodermal syndrome
Efficacy of the pharmacologic chaperone migalastat in a subset of male patients with the classic phenotype of Fabry disease and migalastat-amenable variants: data from the phase 3 randomized, multicen
Correction: Effectiveness of plasma lyso-Gb3 as a biomarker for selecting high-risk patients with Fabry disease from multispecialty clinics for genetic analysis
CRISPR/Cas9-targeted enrichment and long-read sequencing of the Fuchs endothelial corneal dystrophy–associated TCF4 triplet repeat
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Impact of NUDT15 genetics on severe thiopurine-related hematotoxicity in patients with European ancestry European ancestry
News
A suite of automated sequence analyses reduces the number of candidate deleterious variants and reveals a difference between probands and unaffected siblings
Best practice guidelines regarding diagnosis and management of patients with type II collagen disorders
Correction: The ARID1B spectrum in 143 patients: from nonsyndromic intellectual disability to Coffin–Siris syndrome
Response to Juang et al.
Xrare: a machine learning method jointly modeling phenotypes and genetic evidence for rare disease diagnosis
Correction: Secondary findings from clinical genomic sequencing: prevalence, patient perspectives, family history assessment, and health-care costs from a multisite study
CYP2D6-guided opioid therapy improves pain control in CYP2D6 intermediate and poor metabolizers: a pragmatic clinical trial
A survey assessing adoption of the ACMG-AMP guidelines for interpreting sequence variants and identification of areas for continued improvement
Untargeted metabolomic profiling reveals multiple pathway perturbations and new clinical biomarkers in urea cycle disorders
ABCA4-associated disease as a model for missing heritability in autosomal recessive disorders: novel noncoding splice, cis-regulatory, structural, and recurrent hypomorphic variants
Pathogenic variants in USP7 cause a neurodevelopmental disorder with speech delays, altered behavior, and neurologic anomalies
Diagnosis and management of glycogen storage diseases type VI and IX: a clinical practice resource of the American College of Medical Genetics and Genomics (ACMG)
Next-generation sequencing of 32 genes associated with hereditary aortopathies and related disorders of connective tissue in a cohort of 199 patients
Constructing identities: the implications of DTC ancestry testing for tribal communities
Fabry disease cardiac variant IVS4+919 G>A is associated with multiple cardiac gene variants in patients with severe cardiomyopathy and fatal arrhythmia
Insights into genetics, human biology and disease gleaned from family based genomic studies
Revising the Psychiatric Phenotype of Homocystinuria
Deep-intronic ABCA4 variants explain missing heritability in Stargardt disease and allow correction of splice defects by antisense oligonucleotides
Correction: A new microdeletion syndrome involving TBC1D24, ATP6V0C, and PDPK1 causes epilepsy, microcephaly, and developmental delay
Response to Gammal et al.
TBX6-associated congenital scoliosis (TACS) as a clinically distinguishable subtype of congenital scoliosis: further evidence supporting the compound inheritance and TBX6 gene dosage model
The illusion of polygenic disease risk prediction
Transcription alterations of KCNQ1 associated with imprinted methylation defects in the Beckwith–Wiedemann locus
BOADICEA: a comprehensive breast cancer risk prediction model incorporating genetic and nongenetic risk factors
In This Issue
News
Considerations for pharmacogenomic testing in a health system
Phenylalanine and tyrosine measurements across gestation by tandem mass spectrometer on dried blood spot cards from normal pregnant women
Loss of function of SVBP leads to autosomal recessive intellectual disability, microcephaly, ataxia, and hypotonia
Correction: Biallelic sequence and structural variants in RAX2 are a novel cause for autosomal recessive inherited retinal disease
45,X mosaicism in a population-based biobank: implications for Turner syndrome
Biallelic germline nonsense variant of MLH3 underlies polyposis predisposition
Marfan syndrome: improved clinical history results in expanded natural history
Response to Prakash et al.
Cancer communication research in the era of genomics and precision medicine: a scoping review
A CRISPR focus on attitudes and beliefs toward somatic genome editing from stakeholders within the sickle cell disease community
Patient re-contact after revision of genomic test results: points to consider—a statement of the American College of Medical Genetics and Genomics (ACMG)
Microdeletions excluding YWHAE and PAFAH1B1 cause a unique leukoencephalopathy: further delineation of the 17p13.3 microdeletion spectrum
Next-generation phenotyping using computer vision algorithms in rare genomic neurodevelopmental disorders
Estimating the burden and economic impact of pediatric genetic disease
Ethnic identity and engagement with genome sequencing research
Characteristics and evaluation outcomes of genomics curricula for health professional students: a systematic literature review
2.5 years’ experience of GeneMatcher data-sharing: a powerful tool for identifying new genes responsible for rare diseases
Detection of structural variation using target captured next-generation sequencing data for genetic diagnostic testing
Response to Evans et al.
Evidence- and consensus-based recommendations for the use of pegvaliase in adults with phenylketonuria
Protecting trust in medical genetics in the new era of forensics
27 years of prenatal diagnosis for Huntington disease in the United Kingdom
Are women with pathogenic variants in PMS2 and MSH6 really at high lifetime risk of breast cancer?
Development of a newborn screening tool based on bivariate normal limits: using psychosine and galactocerebrosidase determination on dried blood spots to predict Krabbe disease
Atypical cerebral palsy: genomics analysis enables precision medicine
A somatic activating NRAS variant associated with kaposiform lymphangiomatosis
Identifying the deficiencies of current diagnostic criteria for neurofibromatosis 2 using databases of 2777 individuals with molecular testing
Determining the clinical validity of hereditary colorectal cancer and polyposis susceptibility genes using the Clinical Genome Resource Clinical Validity Framework
Pathogenic TERT promoter variants in telomere diseases
Lessons learned from the DFNA37 gene discovery odyssey
ClinPhen extracts and prioritizes patient phenotypes directly from medical records to expedite genetic disease diagnosis
On CALFAN syndrome: report of a patient with a novel variant in SCYL1 gene and recurrent respiratory failure
Secondary research uses of residual newborn screening dried bloodspots: a scoping review
Response to Spagnoli et al.
Analysis of 51 proposed hypertrophic cardiomyopathy genes from genome sequencing data in sarcomere negative cases has negligible diagnostic yield
Clinical validity assessment of genes frequently tested on hereditary breast and ovarian cancer susceptibility sequencing panels
Genome sequencing identifies multiple deleterious variants in autism patients with more severe phenotypes
A functional assay–based procedure to classify mismatch repair gene variants in Lynch syndrome
Lessons learned from two decades of BRCA1 and BRCA2 genetic testing: the evolution of data sharing and variant classification
Frequency of de novo variants and parental mosaicism in vascular Ehlers–Danlos syndrome
Personalized medicine: paradigm shift or revolution
RNA sequencing solved the most common but unrecognized NEB pathogenic variant in Japanese nemaline myopathy
Experiences and perspectives on the return of secondary findings among genetic epidemiologists
Qualitative study of system-level factors related to genomic implementation
Type IV galactosemia
Correction: Putting genome-wide sequencing in neonates into perspective
Haploseek: a 24-hour all-in-one method for preimplantation genetic diagnosis (PGD) of monogenic disease and aneuploidy
Trajectory of exonic variant discovery in a large clinical population: implications for variant curation
Correction: Homozygosity mapping provides supporting evidence of pathogenicity in recessive Mendelian disease
Genetics in Medicine at Twenty
Deficiency of the human cysteine protease inhibitor cystatin M/E causes hypotrichosis and dry skin
Nonimmune hydrops fetalis: identifying the underlying genetic etiology
Genomic mosaicism in the pathogenesis and inheritance of a Rett syndrome cohort
Foundation of the Newborn Screening Translational Research Network and its tools for research
eKLIPse: a sensitive tool for the detection and quantification of mitochondrial DNA deletions from next-generation sequencing data
Knowledge, motivations, expectations, and traits of an African, African-American, and Afro-Caribbean sequencing cohort and comparisons to the original ClinSeq® cohort
Disparities in genetic services utilization in a random sample of young breast cancer survivors
Increasing evidence of combinatory variant effects calls for revised classification of low-penetrance alleles
Stakeholders’ views on the value of outcomes from clinical genetic and genomic interventions
Improved diagnostics by exome sequencing following raw data reevaluation by clinical geneticists involved in the medical care of the individuals tested
Biallelic sequence and structural variants in RAX2 are a novel cause for autosomal recessive inherited retinal disease
Population data improves variant interpretation in autosomal dominant polycystic kidney disease
Giving adolescents a voice: the types of genetic information adolescents choose to learn and why
Outcomes of 92 patient-driven family studies for reclassification of variants of uncertain significance
A logic model for precision medicine implementation informed by stakeholder views and implementation science
The phenotypic spectrum of WWOX-related disorders: 20 additional cases of WOREE syndrome and review of the literature
The ARID1B spectrum in 143 patients: from nonsyndromic intellectual disability to Coffin–Siris syndrome
A framework to build capacity for a reflex-testing program for Lynch syndrome
Biallelic GALM pathogenic variants cause a novel type of galactosemia
Carrier screening “within the panel”
Response to Suthers and Mina
Psychological adaptation to molecular autopsy findings following sudden cardiac death in the young
Trends over 42 years in the Adult Medical Genetics Clinic at the University of Washington
Comparing ethnicity-based and expanded carrier screening methods at a single fertility center reveals significant differences in carrier rates and carrier couple rates
Translating genotype data of 44,000 biobank participants into clinical pharmacogenetic recommendations: challenges and solutions
Correction: TANGO2: expanding the clinical phenotype and spectrum of pathogenic variants
Clinical utility of expanded carrier screening: results-guided actionability and outcomes
Correction: Mapping the genomic landscape of inherited retinal disease genes prioritizes genes prone to coding and noncoding copy-number variations
Pitfalls of clinical exome and gene panel testing: alternative transcripts
Variant data sharing by clinical laboratories through public databases: consent, privacy and further contact for research policies
Response to Hannah-Shmouni and Stratakis
Growth hormone excess in neurofibromatosis 1
Homozygosity mapping provides supporting evidence of pathogenicity in recessive Mendelian disease
Standard operating procedure for somatic variant refinement of sequencing data with paired tumor and normal samples
Putting genome-wide sequencing in neonates into perspective
Copy-number variants in clinical genome sequencing: deployment and interpretation for rare and undiagnosed disease
Variants affecting diverse domains of MEPE are associated with two distinct bone disorders, a craniofacial bone defect and otosclerosis
Response to Esplin et al.
Identification of clinically actionable variants from genome sequencing of families with congenital heart disease
De novo missense variants in RAC3 cause a novel neurodevelopmental syndrome
Secondary findings from clinical genomic sequencing: prevalence, patient perspectives, family history assessment, and health-care costs from a multisite study
Molecular autopsy by trio exome sequencing (ES) and postmortem examination in fetuses and neonates with prenatally identified structural anomalies
Long-term follow-up in PMM2-CDG: are we ready to start treatment trials?
Correction: Expanding the clinical phenotype of individuals with a 3-bp in-frame deletion of the NF1 gene (c.2970_2972del): an update of genotype–phenotype correlation
Correction: IQSEC2-related encephalopathy in males and females: a comparative study including 37 novel patients
Functional analysis of DES-p.L398P and RBM20-p.R636C
Response to Brodehl et al.
Correction: The landscape of epilepsy-related GATOR1 variants
Lessons from the premature adoption of preimplantation embryo testing
Longitudinal growth curves for children with classical osteogenesis imperfecta (types III and IV) caused by structural pathogenic variants in type I collagen
Recall by genotype and cascade screening for familial hypercholesterolemia in a population-based biobank from Estonia
Safety and efficacy of low-dose sirolimus in the PIK3CA-related overgrowth spectrum
Sharing data for future research'engaging participants' views about data governance beyond the original project: a DIRECT Study
Opening the ̏black box̋ of informed consent appointments for genome sequencing: a multisite observational study
The global prevalence of Wilson disease from next-generation sequencing data
TANGO2: expanding the clinical phenotype and spectrum of pathogenic variants
Secondary findings on virtual panels: opportunities, challenges, and potential for preventive medicine
Splice-altering variant in COL11A1 as a cause of nonsyndromic hearing loss DFNA37
Identification of novel loci for pediatric cholestatic liver disease defined by KIF12, PPM1F, USP53, LSR, and WDR83OS pathogenic variants
Autozygome and high throughput confirmation of disease genes candidacy
The evolving landscape of expanded carrier screening: challenges and opportunities
Population-based genetic testing of asymptomatic women for breast and ovarian cancer susceptibility
A new clinical screening strategy and prevalence estimation for glucokinase variant-induced diabetes in an adult Chinese population Chinese population
A new microdeletion syndrome involving TBC1D24, ATP6V0C, and PDPK1 causes epilepsy, microcephaly, and developmental delay
KAT6A Syndrome: genotype–phenotype correlation in 76 patients with pathogenic KAT6A variants
Direct health-care costs for children diagnosed with genetic diseases are significantly higher than for children with other chronic diseases
Factors influencing NCGENES research participants- requests for non-medically actionable secondary findings
Correction: The effect of NOTCH3 pathogenic variant position on CADASIL disease severity: NOTCH3 EGFr 1–6 pathogenic variant are associated with a more severe phenotype and lower survival compared wit
Haploinsufficiency of vascular endothelial growth factor related signaling genes is associated with tetralogy of Fallot
Correction: The clinical spectrum and natural history of early-onset diseases due to DNA polymerase gamma mutations
ATG7 and ATG9A loss-of-function variants trigger autophagy impairment and ovarian failure
CFTR variants and renal abnormalities in males with congenital unilateral absence of the vas deferens (CUAVD): a systematic review and meta-analysis of observational studies
Genomic and phenotypic delineation of congenital microcephaly
An immune tolerance approach using transient low-dose methotrexate in the ERT-naïve setting of patients treated with a therapeutic protein: experience in infantile-onset Pompe disease
Developing a genetic services assessment tool to inform quality improvement efforts in state genetic service delivery
Misattributed parentage as an unanticipated finding during exome/genome sequencing: current clinical laboratory practices and an opportunity for standardization
Correction: Effectiveness of plasma lyso-Gb3 as a biomarker for selecting high-risk patients with Fabry disease from multispecialty clinics for genetic analysis
Use of a novel computerized decision aid for aneuploidy screening: a randomized controlled trial
Combining newborn metabolic and DNA analysis for second-tier testing of methylmalonic acidemia
Frequency and signature of somatic variants in 1461 human brain exomes
Increasing genomic literacy among adolescents
IQSEC2-related encephalopathy in males and females: a comparative study including 37 novel patients
The physician–patient relationship in the age of precision medicine
Correction: Arterial tortuosity syndrome: 40 new families and literature review
Rare variants in the genetic background modulate cognitive and developmental phenotypes in individuals carrying disease-associated variants
Low-level parental mosaicism affects the recurrence risk of holoprosencephaly
HLBS-PopOmics: an online knowledge base to accelerate dissemination and implementation of research advances in population genomics to reduce the burden of heart, lung, blood, and sleep disorders
Correction: Effectiveness of plasma lyso-Gb3 as a biomarker for selecting high-risk patients with Fabry disease from multispecialty clinics for genetic analysis
Expanding the clinical phenotype of individuals with a 3-bp in-frame deletion of the NF1 gene (c.2970_2972del): an update of genotype–phenotype correlation
Development of Clinical Domain Working Groups for the Clinical Genome Resource (ClinGen): lessons learned and plans for the future
Mosaic Turner syndrome shows reduced penetrance in an adult population study
Correction to: Accelerating evidence gathering and approval of precision medicine therapies: the FDA takes aim at rare mutations
Parental interest in genomic sequencing of newborns: enrollment experience from the BabySeq Project
De novo truncating variants in the intronless IRF2BPL are responsible for developmental epileptic encephalopathy
Clinical exome sequencing reveals locus heterogeneity and phenotypic variability of cohesinopathies
Correction: A homozygous FANCM frameshift pathogenic variant causes male infertility
Correction: Does genomic sequencing early in the diagnostic trajectory make a difference? A follow-up study of clinical outcomes and cost-effectiveness
Correction: Opportunities to implement a sustainable genomic medicine program: lessons learned from the IGNITE Network
Correction to: The landscape of epilepsy-related GATOR1 variants
Noncardiac genetic predisposition in sudden infant death syndrome
Correction: The Gen-Equip Project: evaluation and impact of genetics e-learning resources for primary care in six European languages
Correction: DNA breakpoint assay reveals a majority of gross duplications occur in tandem reducing VUS classifications in breast cancer predisposition genes
Correction: “Congenital hyperinsulinism as the presenting feature of Kabuki syndrome: clinical and molecular characterization of 10 affected individuals”
Development of an evidence-based algorithm that optimizes sensitivity and specificity in ES-based diagnostics of a clinically heterogeneous patient population
The landscape of epilepsy-related GATOR1 variants
Paradigm shifts in newborn screening?
Including ELSI research questions in newborn screening pilot studies
Genomic sequencing in acutely ill infants: what will it take to demonstrate clinical value?
The New York pilot newborn screening program for lysosomal storage diseases: Report of the First 65,000 Infants
Analysis of fetal DNA in maternal plasma with markers designed for forensic DNA mixture resolution
Contribution of noncoding pathogenic variants to RPGRIP1-mediated inherited retinal degeneration
DNA breakpoint assay reveals a majority of gross duplications occur in tandem reducing VUS classifications in breast cancer predisposition genes
Disease and subtype specific signatures enable precise diagnosis of the mucopolysaccharidoses
Metabolome-guided genomics to identify pathogenic variants in isocitrate dehydrogenase, fumarate hydratase, and succinate dehydrogenase genes in pheochromocytoma and paraganglioma
Correction: Comparative assessment of gene-specific variant distribution in prenatal and postnatal cohorts tested for Noonan syndrome and related conditions
Response to Biesecker
The Gen-Equip Project: evaluation and impact of genetics e-learning resources for primary care in six European languages
The effect of NOTCH3 pathogenic variant position on CADASIL disease severity: NOTCH3 EGFr 1–6 pathogenic variant are associated with a more severe phenotype and lower survival compared with EGFr 7–34
Response to Mendelsohn and Sabbadini
Clinical genome sequencing in an unbiased pediatric cohort
Phrank measures phenotype sets similarity to greatly improve Mendelian diagnostic disease prioritization
Exome sequencing in congenital ataxia identifies two new candidate genes and highlights a pathophysiological link between some congenital ataxias and early infantile epileptic encephalopathies
Direct-to-consumer raw genetic data and third-party interpretation services: more burden than bargain?
Allelic phenotype values: a model for genotype-based phenotype prediction in phenylketonuria
Opportunities to implement a sustainable genomic medicine program: lessons learned from the IGNITE Network
microRNAs as biomarkers in Pompe disease
The functional impact of variants of uncertain significance in BRCA2
Private payer coverage policies for exome sequencing (ES) in pediatric patients: trends over time and analysis of evidence cited
Health behaviors among unaffected participants following receipt of variants of uncertain significance in cardiomyopathy-associated genes
Developing a conceptual, reproducible, rubric-based approach to consent and result disclosure for genetic testing by clinicians with minimal genetics background
Accelerating evidence gathering and approval of precision medicine therapies: the FDA takes aim at rare mutations
Growth characteristics in individuals with osteogenesis imperfecta in North America: results from a multicenter study
Normative and conceptual ELSI research: what it is, and why it’s important
Noonan syndrome associated with growth hormone deficiency with biallelic LZTR1 variants
Response to Nakaguma et al.
Beyond the panel: preconception screening in consanguineous couples using the TruSight One “clinical exome”
Genome sequencing as a first-line genetic test in familial dilated cardiomyopathy
Germline cancer susceptibility gene variants, somatic second hits, and survival outcomes in patients with resected pancreatic cancer
Population genomics in South East Asia captures unexpectedly high carrier frequency for treatable inherited disorders
Advocating for the consumer: clinical confirmation of all direct-to-consumer raw data alterations remains critical
Addressing the accuracy of direct-to-consumer genetic testing
Genomic characterization of the RH locus detects complex and novel structural variation in multi-ethnic cohorts
MOVR—NeuroMuscular ObserVational Research, a unified data hub for neuromuscular diseases
Gene modification therapies: views of parents of people with Down syndrome
Clinical and genetic spectrum of a large cohort of children with epilepsy in China
TRPV1 variants impair intracellular Ca2+ signaling and may confer susceptibility to malignant hyperthermia
Bioinformatics for medical students: a 5-year experience using OMIM® in medical student education
MYLK pathogenic variants aortic disease presentation, pregnancy risk, and characterization of pathogenic missense variants
Clinical implications of systematic phenotyping and exome sequencing in patients with primary antibody deficiency
Big data phenotyping in rare diseases: some ethical issues
A recurrent de novo missense pathogenic variant in SMARCB1 causes severe intellectual disability and choroid plexus hyperplasia with resultant hydrocephalus
Evaluating the role of public health in implementation of genomics-related recommendations: a case study of hereditary cancers using the CDC Science Impact Framework
Comparative assessment of gene-specific variant distribution in prenatal and postnatal cohorts tested for Noonan syndrome and related conditions
A comprehensive iterative approach is highly effective in diagnosing individuals who are exome negative
Congenital hyperinsulinism as the presenting feature of Kabuki syndrome: clinical and molecular characterization of 10 affected individuals
A genotype-specific surgical approach for patients with Pfeiffer syndrome due to W290C pathogenic variant in FGFR2 is associated with improved developmental outcomes and reduced mortality
Targeted exome analysis identifies the genetic basis of disease in over 50% of patients with a wide range of ataxia-related phenotypes
Development of a consent resource for genomic data sharing in the clinical setting
First, do no harm: direct-to-consumer genetic testing
From exome analysis in idiopathic azoospermia to the identification of a high-risk subgroup for occult Fanconi anemia
When genomic medicine reveals misattributed genetic relationships—the debate about disclosure revisited
Physicians’ perspectives on receiving unsolicited genomic results
A homozygous FANCM frameshift pathogenic variant causes male infertility
Understanding the potential of state-based public health genomics programs to mitigate disparities in access to clinical genetic services
Prevalence and properties of intragenic copy-number variation in Mendelian disease genes
Genome-wide sequencing in acutely ill infants: genomic medicine’s critical application?
Evaluation of polygenic risk models using multiple performance measures: a critical assessment of discordant results
Evaluating the pathogenic potential of genes with de novo variants in epileptic encephalopathies
Novel phenotype–disease matching tool for rare genetic diseases
ZP2 pathogenic variants cause in vitro fertilization failure and female infertility
Introducing medical genetics services in Ethiopia using the MiGene Family History App
De novo truncating variants in WHSC1 recapitulate the Wolf–Hirschhorn (4p16.3 microdeletion) syndrome phenotype
A gene-centric strategy for identifying disease-causing rare variants in dilated cardiomyopathy
“I don’t want to be Henrietta Lacks”: diverse patient perspectives on donating biospecimens for precision medicine research
Comprehensive annotation of BRCA1 and BRCA2 missense variants by functionally validated sequence-based computational prediction models
A proposed nosology of inborn errors of metabolism
Implementation, adoption, and utility of family health history risk assessment in diverse care settings: evaluating implementation processes and impact with an implementation framework
Identification of lysosomal and extralysosomal globotriaosylceramide (Gb3) accumulations before the occurrence of typical pathological changes in the endomyocardial biopsies of Fabry disease patients
A cross-sectional quantitative analysis of the natural history of free sialic acid storage disease—an ultra-orphan multisystemic lysosomal storage disorder
Returning negative results to individuals in a genomic screening program: lessons learned
The impact of variant classification on the clinical management of hereditary cancer syndromes
Secondary findings from next-generation sequencing: what does actionable in childhood really mean?
Aminoacyl-tRNA synthetase deficiencies in search of common themes
Macrocytic anemia in Lesch–Nyhan disease and its variants
Stargazer: a software tool for calling star alleles from next-generation sequencing data using CYP2D6 as a model
Written pretest information and germline BRCA1/2 pathogenic variant testing in unselected breast cancer patients: predictors of testing uptake
Defining the diagnostic effectiveness of genes for inclusion in panels: the experience of two decades of genetic testing for hypertrophic cardiomyopathy at a single center
Cervical spinal cord compression in infants with achondroplasia: should neuroimaging be routine?
Pharmacogenetic testing in the Veterans Health Administration (VHA): policy recommendations from the VHA Clinical Pharmacogenetics Subcommittee
Secondary findings in exome slices, virtual panels, and anticipatory sequencing
Response to Roberts et al. 2018: is breast cancer truly caused by MSH6 and PMS2 variants or is it simply due to a high prevalence of these variants in the population?
Response to ten Broeke
Is universal tumor testing for Lynch syndrome cost-effective? It depends!
Does genomic sequencing early in the diagnostic trajectory make a difference? A follow-up study of clinical outcomes and cost-effectiveness
Clinical Application of Genome and Exome Sequencing as a Diagnostic Tool for Pediatric Patients: a Scoping Review of the Literature
Universal screening of Lynch syndrome is ready for implementation
Validation of Version 3.0 of the Breast Cancer Genetics Referral Screening Tool (B-RST™)
Determining pathogenicity of genetic variants in hypertrophic cardiomyopathy: importance of periodic reassessment
Effectiveness of plasma lyso-Gb3 as a biomarker for selecting high-risk patients with Fabry disease from multispecialty clinics for genetic analysis
The current state of funded NIH grants in implementation science in genomic medicine: a portfolio analysis
Preemptive pharmacogenetic testing: exploring the knowledge and perspectives of US payers
Medical genetics and genomics education and its impact on genomic literacy of the clinical workforce
Loss-of-function mutations in TDRD7 lead to a rare novel syndrome combining congenital cataract and nonobstructive azoospermia in humans