Genetics in Medicine - 2020

340 articles | Last updated: 2025-12-03 14:12:56
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T A T A T A T A
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Correction: A longitudinal footprint of genetic epilepsies using automated electronic medical record interpretation
Alaska Native genomic research: perspectives from Alaska Native leaders, federal staff, and biomedical researchers
Correction: KAT6A Syndrome: genotype–phenotype correlation in 76 patients with pathogenic KAT6A variants
Expanding the clinical and genetic spectrum of CAD deficiency: an epileptic encephalopathy treatable with uridine supplementation
Health-care practitioners’ preferences for the return of secondary findings from next-generation sequencing: a discrete choice experiment
Limited diagnostic impact of duplications <1 Mb of uncertain clinical significance: a 10-year retrospective analysis of reporting practices at the Mayo Clinic
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Airmen and health-care providers’ attitudes toward the use of genomic sequencing in the US Air Force: findings from the MilSeq Project
Genome-wide noninvasive prenatal screening for carriers of balanced reciprocal translocations
An Anti-Racism Toolkit for the Genetics Educator
Role of POLE and POLD1 in familial cancer
Constitutional mismatch repair deficiency is the diagnosis in 0.41% of pathogenic NF1/SPRED1 variant negative children suspected of sporadic neurofibromatosis type 1
Considerations in assessing germline variant pathogenicity using cosegregation analysis
A longitudinal footprint of genetic epilepsies using automated electronic medical record interpretation
A cost-effectiveness analysis of genomic sequencing in a prospective versus historical cohort of complex pediatric patients
Genotype–phenotype correlations in recessive titinopathies
DYNC2H1 hypomorphic or retina-predominant variants cause nonsyndromic retinal degeneration
Genetic dissection of spermatogenic arrest through exome analysis: clinical implications for the management of azoospermic men
Clinical, molecular, and biochemical delineation of asparagine synthetase deficiency in Saudi cohort
Treatment of mucopolysaccharidosis type II (Hunter syndrome): a Delphi derived practice resource of the American College of Medical Genetics and Genomics (ACMG)
Frequency of the loss of CAA interruption in the HTT CAG tract and implications for Huntington disease in the reduced penetrance range
Tumor detection rates in screening of individuals with SDHx-related hereditary paraganglioma–pheochromocytoma syndrome
Assessing non-Mendelian inheritance in inherited axonopathies
Application of exome sequencing for prenatal diagnosis: a rapid scoping review
Correction: Evaluating the extent of reusability of CYP2C19 genotype data among patients genotyped for antiplatelet therapy selection
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Clinical, neuropathological, and genetic characterization of STUB1 variants in cerebellar ataxias: a frequent cause of predominant cognitive impairment
Correction: Meta-analysis and multidisciplinary consensus statement: exome sequencing is a first-tier clinical diagnostic test for individuals with neurodevelopmental disorders
Pathogenic mosaic variants in congenital hypogonadotropic hypogonadism
Correction: Opportunities to implement a sustainable genomic medicine program: lessons learned from the IGNITE Network
Correction: Developing a common framework for evaluating the implementation of genomic medicine interventions in clinical care: the IGNITE Network’s Common Measures Working Group
A genomics approach to male infertility
Parental experiences of ultrarapid genomic testing for their critically unwell infants and children
Likely damaging de novo variants in congenital diaphragmatic hernia patients are associated with worse clinical outcomes
Genotypic diversity and phenotypic spectrum of infantile liver failure syndrome type 1 due to variants in LARS1
De novo SMARCA2 variants clustered outside the helicase domain cause a new recognizable syndrome with intellectual disability and blepharophimosis distinct from Nicolaides–Baraitser syndrome
Correction: Cancer risks by gene, age, and gender in 6350 carriers of pathogenic mismatch repair variants: findings from the Prospective Lynch Syndrome Database
Patient reactions to receiving negative genomic screening results by mail
Participant choices for return of genomic results in the eMERGE Network
Low-level parental somatic mosaic SNVs in exomes from a large cohort of trios with diverse suspected Mendelian conditions
Polygenic risk scores and breast and epithelial ovarian cancer risks for carriers of BRCA1 and BRCA2 pathogenic variants
The multiple faces of urinary glucose tetrasaccharide as biomarker for patients with hepatic glycogen storage diseases
Response to Heiner-Fokkema et al.
Reduced penetrance of pathogenic ACMG variants in a deeply phenotyped cohort study and evaluation of ClinVar classification over time
Evaluating the extent of reusability of CYP2C19 genotype data among patients genotyped for antiplatelet therapy selection
A six-attribute classification of geneticmosaicism
Tracing the mutated HTT and haplotype of the African ancestor who spread Huntington disease into the Middle East African ancestor
Correction: SMAD6 variants in craniosynostosis: genotype and phenotype evaluation
Improved structural variant interpretation for hereditary cancer susceptibility using long-read sequencing
Addendum: American College of Medical Genetics guideline on the cytogenetic evaluation of the individual with developmental delay or mental retardation
Validation of the BOADICEA model and a 313-variant polygenic risk score for breast cancer risk prediction in a Dutch prospective cohort
Correction: The composition and capacity of the clinical genetics workforce in high-income countries: a scoping review
Educating military primary health-care providers in genomic medicine: lessons learned from the MilSeq Project
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Teaching clinicians practical genomic medicine: 7 years’ experience in a tertiary care center
Genetic ancestry analysis on >93,000 individuals undergoing expanded carrier screening reveals limitations of ethnicity-based medical guidelines Genetic ancestry; ethnicity-based medical guidelines
Response to Ferket et al.
Medically actionable pathogenic variants in a population of 13,131 healthy elderly individuals
Correspondence on: “Discrepancy in Spinal Muscular Atrophy Incidence findings in newborn screening programs: the influence of carrier screening?” by Kay et al
Economic value of exome sequencing for suspected monogenic disorders
Patient and family social media use surrounding a novel treatment for a rare genetic disease: a qualitative interview study
Message from ACMG President: overcoming disparities
A molecular basis for neurofibroma-associated skeletal manifestations in NF1
Clinical outcomes of a genomic screening program for actionable genetic conditions
Correction: Addendum: ACMG Practice Guideline: lack of evidence for MTHFR polymorphism testing
BLOC1S5 pathogenic variants cause a new type of Hermansky–Pudlak syndrome
Genetic testing and results disclosure in diverse populations: what does it take?
Prenatal exome sequencing in 65 fetuses with abnormality of the corpus callosum: contribution to further diagnostic delineation
CNVs cause autosomal recessive genetic diseases with or without involvement of SNV/indels
The composition and capacity of the clinical genetics workforce in high-income countries: a scoping review
Correction: Alternative mRNA splicing can attenuate the pathogenicity of presumed loss-of-function variants in BRCA2
Classical-like Ehlers–Danlos syndrome: a clinical description of 20 newly identified individuals with evidence of tissue fragility
Late morbidity and mortality in adult survivors of childhood glioma with neurofibromatosis type 1: report from the Childhood Cancer Survivor Study
Frequency of genomic secondary findings among 21,915 eMERGE network participants
Ethical conflicts in translational genetic research: lessons learned from the eMERGE-III experience
Correction: Phenotate: crowdsourcing phenotype annotations as exercises in undergraduate classes
Correction: An approach to integrating exome sequencing for fetal structural anomalies into clinical practice
What’s in a name? Issues to consider when naming Mendelian disorders
Training the next generation of genomic medicine providers: trends in medical education and national assessment
Genetics and pediatric hospital admissions, 1985 to 2017
Sequential targeted exome sequencing of 1001 patients affected by unexplained limb-girdle weakness
The limited use of US residual newborn screening dried bloodspots for health disparity research
Addendum: ACMG Practice Guideline: lack of evidence for MTHFR polymorphism testing
Toward newborn screening of cerebrotendinous xanthomatosis: results of a biomarker research study using 32,000 newborn dried blood spots
Cardiac involvement in classical or hypermobile Ehlers–Danlos syndrome is uncommon
Addendum: Statement on nutritional supplements and piracetam for children with Down syndrome
Addendum: Array-based technology and recommendations for utilization in medical genetics practice for detection of chromosomal abnormalities
Societal considerations in host genome testing for COVID-19
COVID-19’s Impact on Genetics at One Medical Center in New York
SMAD6 variants in craniosynostosis: genotype and phenotype evaluation
Correction: Biallelic GALM pathogenic variants cause a novel type of galactosemia
Revealing hidden genetic diagnoses in the ocular anterior segment disorders
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A rapid solubility assay of protein domain misfolding for pathogenicity assessment of rare DNA sequence variants
Phenotype expansion of heterozygous FOXC1 pathogenic variants toward involvement of congenital anomalies of the kidneys and urinary tract (CAKUT)
Automated syndrome diagnosis by three-dimensional facial imaging
Morbidity in 47,XYY syndrome: a nationwide epidemiological study of hospital diagnoses and medication use
The interface of genomic information with the electronic health record: a points to consider statement of the American College of Medical Genetics and Genomics (ACMG)
Correction: Genome and RNA sequencing in patients with methylmalonic aciduria of unknown cause
Correction: Clinical experience with carrier screening in a general population: support for a comprehensive pan-ethnic approach
Confirmation of risk of cancer in blepharocheilodontic syndrome
TSC2 pathogenic variants are predictive of severe clinical manifestations in TSC infants: results of the EPISTOP study
How can Australia integrate routine genetic sequencing in oncology: a qualitative study through an implementation science lens
Cell-based analysis of CAD variants identifies individuals likely to benefit from uridine therapy
Correction: The genomic and clinical landscape of fetal akinesia
Integrated analysis of metabolomic profiling and exome data supplements sequence variant interpretation, classification, and diagnosis
Diagnosing hereditary cancer predisposition in men with prostate cancer
Response to Bai et al.
Implementation of population-based newborn screening reveals low incidence of spinal muscular atrophy
Comment on the criteria for interpretation of mitochondrial tRNA variants
Further delineation of the clinical spectrum of KAT6B disorders and allelic series of pathogenic variants
Patient experience of uncertainty in cancer genomics: a systematic review
Genetic, structural, and functional characterization of POLE polymerase proofreading variants allows cancer risk prediction
Overcoming the challenges associated with universal screening for Lynch syndrome in colorectal and endometrial cancer
Ensuring continuity of care for children with inherited metabolic diseases at the time of COVID-19: the experience of a metabolic unit in Italy
Response to Benusiglio et al.
CFTR variant testing: a technical standard of the American College of Medical Genetics and Genomics (ACMG)
Points to consider when assessing relationships (or suspecting misattributed relationships) during family-based clinical genomic testing: a statement of the American College of Medical Genetics and Ge
The leadership behaviors needed to implement clinical genomics at scale: a qualitative study
Disease expression in juvenile polyposis syndrome: a retrospective survey on a cohort of 221 European patients and comparison with a literature-derived cohort of 473 SMAD4/BMPR1A pathogenic variant ca European patients
Matching clinical and genetic diagnoses in autosomal dominant polycystic kidney disease reveals novel phenocopies and potential candidate genes
COVID-19 outcomes and the human genome
Alternative mRNA splicing can attenuate the pathogenicity of presumed loss-of-function variants in BRCA2
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Longitudinal follow-up after telephone disclosure in the randomized COGENT study
Phenotypic spectrum and transcriptomic profile associated with germline variants in TRAF7
Returning genomic results in a Federally Qualified Health Center: the intersection of precision medicine and social determinants of health
The personal utility and uptake of genomic sequencing in pediatric and adult conditions: eliciting societal preferences with three discrete choice experiments
Biases in arginine codon usage correlate with genetic disease risk
Phenotate: crowdsourcing phenotype annotations as exercises in undergraduate classes
Clinical experience with carrier screening in a general population: support for a comprehensive pan-ethnic approach
De novo variants of NR4A2 are associated with neurodevelopmental disorder and epilepsy
Alternative transcripts in variant interpretation: the potential for missed diagnoses and misdiagnoses
Loss-of-function variants in C3ORF52 result in localized autosomal recessive hypotrichosis
Exome sequencing compared with standard genetic tests for critically ill infants with suspected genetic conditions
Positive impact of genetic counseling assistants on genetic counseling efficiency, patient volume, and cost in a cancer genetics clinic
Response to Maya et al.
Plasma cell-free DNA variant analysis compared with methylated DNA analysis in renal cell carcinoma
The prevalence of genetic diagnoses in fetuses with severe congenital heart defects
High-frequency low-penetrance copy-number variant classification: should we revise the existing guidelines?
DLG2 variants in patients with pubertal disorders
Points to consider for reporting of germline variation in patients undergoing tumor testing: a statement of the American College of Medical Genetics and Genomics (ACMG)
A highly multiplexed biochemical assay for analytes in dried blood spots: application to newborn screening and diagnosis of lysosomal storage disorders and other inborn errors of metabolism
Resolving the dark matter of ABCA4 for 1054 Stargardt disease probands through integrated genomics and transcriptomics
Mainstreaming genetics and genomics: a systematic review of the barriers and facilitators for nurses and physicians in secondary and tertiary care
The diagnostic utility of genome sequencing in a pediatric cohort with suspected mitochondrial disease
Curaçao diagnostic criteria for hereditary hemorrhagic telangiectasia is highly predictive of a pathogenic variant in ENG or ACVRL1 (HHT1 and HHT2)
Diagnostic testing for uniparental disomy: a points to consider statement from the American College of Medical Genetics and Genomics (ACMG)
Electronic health record phenotypes associated with genetically regulated expression of CFTR and application to cystic fibrosis
The landscape of pharmacogenetic testing in a US managed care population
X-linked diseases: susceptible females
Correction: Interpretation of mitochondrial tRNA variants
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Mapping RNA splicing variations in clinically accessible and nonaccessible tissues to facilitate Mendelian disease diagnosis using RNA-seq
Correction: Blood RNA analysis can increase clinical diagnostic rate and resolve variants of uncertain significance
Correction: A restricted spectrum of missense KMT2D variants cause a multiple malformations disorder distinct from Kabuki syndrome
Systematic evidence-based review: outcomes from exome and genome sequencing for pediatric patients with congenital anomalies or intellectual disability
Initial experience from a renal genetics clinic demonstrates a distinct role in patient management
Significant Mendelian genetic contribution to pediatric mild-to-moderate hearing loss and its comprehensive diagnostic approach
Re-evaluating the first-tier status of fragile X testing in neurodevelopmental disorders
Correction: Interpretation of mitochondrial tRNA variants
Risk categorization for oversight of laboratory-developed tests for inherited conditions: an updated position statement of the American College of Medical Genetics and Genomics (ACMG)
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Blood RNA analysis can increase clinical diagnostic rate and resolve variants of uncertain significance
Multisystem burden of neurofibromatosis 1 in Denmark: registry- and population-based rates of hospitalizations over the life span
Loss of TNR causes a nonprogressive neurodevelopmental disorder with spasticity and transient opisthotonus
The critical role of psychosine in screening, diagnosis, and monitoring of Krabbe disease
Integrating stakeholder feedback in translational genomics research: an ethnographic analysis of a study protocol’s evolution
Clinical and biochemical improvement with galactose supplementation in SLC35A2-CDG
Regulation of human cerebral cortical development by EXOC7 and EXOC8, components of the exocyst complex, and roles in neural progenitor cell proliferation and survival
Spinal muscular atrophy diagnosis and carrier screening from genome sequencing data
Disparities in BRCA counseling across providers in a diverse population of young breast cancer survivors
Loss-of-function variants in CTNNA1 detected on multigene panel testing in individuals with gastric or breast cancer
The morbid genome of ciliopathies: an update
Correction: GATAD2B-associated neurodevelopmental disorder (GAND): clinical and molecular insights into a NuRD-related disorder
Copy-number variation contributes 9% of pathogenicity in the inherited retinal degenerations
The Cardiac Genome Clinic: implementing genome sequencing in pediatric heart disease
Noninvasive prenatal diagnosis by genome-wide haplotyping of cell-free plasma DNA
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Examination of the genetic factors underlying the cognitive variability associated with neurofibromatosis type 1
Cost-effectiveness of long-term clinical management of BRCA pathogenic variant carriers
Exome sequencing in newborns with congenital deafness as a model for genomic newborn screening: the Baby Beyond Hearing project
An approach to integrating exome sequencing for fetal structural anomalies into clinical practice
Two integrated and highly predictive functional analysis-based procedures for the classification of MSH6 variants in Lynch syndrome
Mobile element insertion detection in 89,874 clinical exomes
Interpretation of mitochondrial tRNA variants
Defining the clinical phenotype of Saul–Wilson syndrome
A restricted spectrum of missense KMT2D variants cause a multiple malformations disorder distinct fromKabuki syndrome
GATAD2B-associatedneurodevelopmental disorder (GAND): clinical and molecular insights into a NuRD-relateddisorder
Assessment of laboratories offering cell-free (cf) DNA screening for Down syndrome: results of the 2018 College of American Pathology External Educational Exercises
The use of fetal exome sequencing in prenatal diagnosis: a points to consider document of the American College of Medical Genetics and Genomics (ACMG)
Systematic misclassification of missense variants in BRCA1 and BRCA2 “coldspots”
Diagnosing Cornelia de Lange syndrome and related neurodevelopmental disorders using RNA sequencing
Higher dosing of alglucosidase alfa improves outcomes in children with Pompe disease: a clinical study and review of the literature
Efficacy and safety of D,L-3-hydroxybutyrate (D,L-3-HB) treatment in multiple acyl-CoA dehydrogenase deficiency
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Clustering of comorbid conditions among women who carry an FMR1 premutation
Assessing relatives’ readiness for hereditary cancer cascade genetic testing
Correction: DYRK1A-related intellectual disability: a syndrome associated with congenital anomalies of the kidney and urinary tract
Parents’ perceptions of personal utility of exome sequencing results
Clinical utility of genetic testing in 201 preschool children with inherited eye disorders
Classification of variants of uncertain significance in BRCA1 and BRCA2 using personal and family history of cancer from individuals in a large hereditary cancer multigene panel testing cohort
Correction: Population screening for BRCA1/BRCA2 founder mutations in Ashkenazi Jews: proactive recruitment compared with self-referral
Prevalence of pathogenic germline cancer risk variants in high-risk urothelial carcinoma
Correction: Adapting ACMG/AMP sequence variant classification guidelines for single-gene copy-number variants
Correction: The CHD4-related syndrome: a comprehensive investigation of the clinical spectrum, genotype–phenotype correlations, and molecular basis
Germline genetic testing for breast cancer: which patients? What genes?
Points to consider: is there evidence to support BRCA1/2 and other inherited breast cancer genetic testing for all breast cancer patients? A statement of the American College of Medical Genetics and G
Response to Potuijt et al.
It all begins with the phenotype
Effectiveness of the Genomics ADvISER decision aid for the selection of secondary findings from genomic sequencing: a randomized clinical trial
Correction: Inferring fetal fractions from read heterozygosity empowers the noninvasive prenatal screening
Laboratory diagnosis of disorders of peroxisomal biogenesis and function: a technical standard of the American College of Medical Genetics and Genomics (ACMG)
The case for implementing sustainable routine, population-level genomic reanalysis
Laird G. Jackson, MD, FACMG
A structured genetics rotation for pediatric residents: an important educational opportunity
Response to Dominguez-Valentin M et al. 2019: Cancer risks by gene, age, and gender in 6350 carriers of pathogenic mismatch repair variants: findings from the Prospective Lynch Syndrome Database
Response to Tolva et al.
Prospective, phenotype-driven selection of critically ill neonates for rapid exome sequencing is associated with high diagnostic yield
De novo CLTC variants are associated with a variable phenotype from mild to severe intellectual disability, microcephaly, hypoplasia of the corpus callosum, and epilepsy
Pediatric Outcomes Data Collection Instrument is a Useful Patient-Reported Outcome Measure for Physical Function in Children with Osteogenesis Imperfecta
Correction: Genetic testing for Parkinson disease: current practice, knowledge, and attitudes among US and Canadian movement disorders specialists
Correction: The Genomics Research and Innovation Network: creating an interoperable, federated, genomics learning system
Unexplained regression in Down syndrome: 35 cases from an international Down syndrome database
Optimizing genetics online resources for diverse readers
When should genomic and exome sequencing be implemented in newborns? A call for an update to newborn screening guidelines
Accurate detection of clinically relevant uniparental disomy from exome sequencing data
Defining clinical subgroups and genotype–phenotype correlations in NBAS-associated disease across 110 patients
Variant interpretation is a component of clinical practice among genetic counselors in multiple specialties
The case for banning heritable genome editing
Diagnostic gene sequencing panels: from design to report—a technical standard of the American College of Medical Genetics and Genomics (ACMG)
Response to Rubanovich et al.
Commentary on the development of the Clinician-reported Genetic testing Utility InDEx (C-GUIDE)
De novo and inherited variants in ZNF292 underlie a neurodevelopmental disorder with features of autism spectrum disorder
Response to Zhang et al.
Detection of iron deficiency in children with Down syndrome
Laboratory screening and diagnosis of open neural tube defects, 2019 revision: a technical standard of the American College of Medical Genetics and Genomics (ACMG)
Recurrent heterozygous PAX6 missense variants cause severe bilateral microphthalmia via predictable effects on DNA–protein interaction
The clinical benefit of genome-wide cfDNA testing cannot be extrapolated from CVS data
Evaluating the Patient-Reported Outcomes Measurement Information System scales in acute intermittent porphyria
Technical standards for the interpretation and reporting of constitutional copy-number variants: a joint consensus recommendation of the American College of Medical Genetics and Genomics (ACMG) and th
Evaluating the role of MLH3 p.Ser1188Ter variant in inherited breast cancer predisposition
Genetic testing for Parkinson disease: current practice, knowledge, and attitudes among US and Canadian movement disorders specialists
The genomic and clinical landscape of fetal akinesia
Response to Sistermans et al.
POLR1B and neural crest cell anomalies in Treacher Collins syndrome type 4
Response to Baertling et al.
Fatal metabolic decompensation in carbonic anhydrase VA deficiency despite early treatment and control of hyperammonemia
Functional characterization of 84 PALB2 variants of uncertain significance
Preconception carrier screening yield: effect of variants of unknown significance in partners of carriers with clinically significant variants
Is there a duty to reinterpret genetic data? The ethical dimensions
The implementation of newborn screening for spinal muscular atrophy: the Australian experience
Exome sequencing: value is in the eye of the beholder
Diagnostic utility of transcriptome sequencing for rare Mendelian diseases
Health-care providers’ perspectives on uncertainty generated by variant forms of newborn screening targets
Correction: Breast cancer in neurofibromatosis 1: survival and risk of contralateral breast cancer in a five country cohort study
Systematic review of the evidence on the cost-effectiveness of pharmacogenomics-guided treatment for cardiovascular diseases
Response to Gomy and Garber
Access to reproductive options after prenatal diagnosis—patient access and physician responsibilities: an updated position statement of the American College of Medical Genetics and Genomics (ACMG)
Widening of the genetic and clinical spectrum of Lamb–Shaffer syndrome, a neurodevelopmental disorder due to SOX5 haploinsufficiency
One step forward, two steps backward
Heme as an initial treatment for severe decompensation in tyrosinemia type 1
Response to Toutain et al.
Response to Neeleman et al.
Type 3 confined placental mosaicisms excluding trisomies 16 are also associated with adverse pregnancy outcomes
Correction: Creating genetic reports that are understood by nonspecialists: a case study
Adapting ACMG/AMP sequence variant classification guidelines for single-gene copy number variants
Therapeutic approaches in Congenital Disorders of Glycosylation (CDG) involving N-linked glycosylation: an update
Clinical utility of exome sequencing in infantile heart failure
Correction: Regional models of genetic services in the United States
Breast cancer in neurofibromatosis 1: survival and risk of contralateral breast cancer in a five country cohort study
Creating genetic reports that are understood by nonspecialists: a case study
Perspectives of US private payers on insurance coverage for pediatric and prenatal exome sequencing: Results of a study from the Program in Prenatal and Pediatric Genomic Sequencing (P3EGS)
Regional models of genetic services in the United States
The pleiotropy associated with de novo variants in CHD4, CNOT3, and SETD5 extends to moyamoya angiopathy
Pathogenic variants in CDC45 on the remaining allele in patients with a chromosome 22q11.2 deletion result in a novel autosomal recessive condition
Psychiatric genomics researchers’ perspectives on best practices for returning results to individual participants
The Genomics Research and Innovation Network: creating an interoperable, federated, genomics learning system
Parents of newborns in the NICU enrolled in genome sequencing research: hopeful, but not naïve
Inferring fetal fractions from read heterozygosity empowers the noninvasive prenatal screening
Prenatal genetic testing for cystic fibrosis: a systematic review of clinical effectiveness and an ethics review
AVADA: toward automated pathogenic variant evidence retrieval directly from the full-text literature
Low-pass genome sequencing versus chromosomal microarray analysis: implementation in prenatal diagnosis
Genome and RNA sequencing in patients with methylmalonic aciduria of unknown cause
The cost trajectory of the diagnostic care pathway for children with suspected genetic disorders
The natural history of infantile mitochondrial DNA depletion syndrome due to RRM2B deficiency
Detection of iron deficiency in children with Down syndrome
Genetics workforce: distribution of genetics services and challenges to health care in California
Who’s on third? Regulation of third-party genetic interpretation services
A clinical guide to hereditary cancer panel testing: evaluation of gene-specific cancer associations and sensitivity of genetic testing criteria in a cohort of 165,000 high-risk patients
Response to Cody
A novel ZRS variant causes preaxial polydactyly type I by increased sonic hedgehog expression in the developing limb bud
Re-evaluation of the classification system for the clinical interpretation of genomic copy number variation
Outcomes in pregnancies with a confined placental mosaicism and implications for prenatal screening using cell-free DNA
The CHD4-related syndrome: a comprehensive investigation of the clinical spectrum, genotype–phenotype correlations, and molecular basis
A genome-first approach to aggregating rare genetic variants in LMNA for association with electronic health record phenotypes
CDKN2A testing and genetic counseling promote reductions in objectively measured sun exposure one year later
Immunopathological manifestations in Kabuki syndrome: a registry study of 177 individuals
A genetic model for multimorbidity in young adults
The development of the Clinician-reported Genetic testing Utility InDEx (C-GUIDE): a novel strategy for measuring the clinical utility of genetic testing
The complete costs of genome sequencing: a microcosting study in cancer and rare diseases from a single center in the United Kingdom
Correction: Sherloc: a comprehensive refinement of the ACMG–AMP variant classification criteria
Challenges in clinical implementation of CYP2D6 genotyping: choice of variants to test affects phenotype determination
Developing interactions with industry in rare diseases: lessons learned and continuing challenges
Outcomes of patient self-referral for the diagnosis of several rare inherited kidney diseases
Pathogenic variants in the DEAH-box RNA helicase DHX37 are a frequent cause of 46,XY gonadal dysgenesis and 46,XY testicular regression syndrome
Cancer risks by gene, age, and gender in 6350 carriers of pathogenic mismatch repair variants: findings from the Prospective Lynch Syndrome Database
Nonrandom occurrence of multiple de novo coding variants in a proband indicates the existence of an oligogenic model in autism
Genomic knowledge in the context of diagnostic exome sequencing: changes over time, persistent subgroup differences, and associations with psychological sequencing outcomes
Return of raw data in genomic testing and research: ownership, partnership, and risk–benefit
A clinical scoring system for congenital contractural arachnodactyly
Frequency and clinical outcomes of CYP2C19 genotype-guided escalation and de-escalation of antiplatelet therapy in a real-world clinical setting
The changing face of clinical genetics service delivery in the era of genomics: a framework for monitoring service delivery and data from a comprehensive metropolitan general genetics service
Thyroid hormone and folinic acid in young children with Down syndrome: the phase 3 ACTHYF trial
Exome sequencing revealed Notch ligand JAG1 as a novel candidate gene for familial exudative vitreoretinopathy
Targeted resequencing of FECH locus reveals that a novel deep intronic pathogenic variant and eQTLs may cause erythropoietic protoporphyria (EPP) through a methylation-dependent mechanism
Incidence of mosaicism in 1055 de novo NF2 cases: much higher than previous estimates with high utility of next-generation sequencing
Using the Delphi method to identify clinicians’ perceived importance of pediatric exome sequencing results
Novel defects in collagen XII and VI expand the mixed myopathy/Ehlers–Danlos syndrome spectrum and lead to variant-specific alterations in the extracellular matrix
Genetic Medicine for Adult Onset Disease
Nucleic Acids, Genes, and Genomes
Laboratory Techniques and the Sequencing Revolution
Pharmacogenetics and Personalized Medicine
Mutations and Genetic Variation
The Application of Genetic Medicine in Childhood