Genetics in Medicine - 2021

369 articles | Last updated: 2025-12-03 14:12:56
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CNV profiles of Chinese pediatric patients with developmental disorders
News
In This Issue
Correction: Clinical utility of genetic testing in 201 preschool children with inherited eye disorders
Correction: Ethical and public health implications of genetic testing for suicide risk: family and survivor perspectives
Correction: Clinical, neuropathological, and genetic characterization of STUB1 variants in cerebellar ataxias: a frequent cause of predominant cognitive impairment
Correction: Low-level mosaicism in tuberous sclerosis complex: prevalence, clinical features, and risk of disease transmission
A retrospective study on the efficacy of prenatal diagnosis for pregnancies at risk of mitochondrial DNA disorders
Autosomal recessive cardiomyopathy and sudden cardiac death associated with variants in MYL3
Real-world integration of genomic data into the electronic health record: the PennChart Genomics Initiative
De novo variants in SNAP25 cause an early-onset developmental and epileptic encephalopathy
Health and economic outcomes of newborn screening for infantile-onset Pompe disease
Correction: Opportunities to implement a sustainable genomic medicine program: lessons learned from the IGNITE Network
Risk-reducing hysterectomy and bilateral salpingo-oophorectomy in female heterozygotes of pathogenic mismatch repair variants: a Prospective Lynch Syndrome Database report
De novo loss-of-function variants in X-linked MED12 are associated with Hardikar syndrome in females
A state-based approach to genomics for rare disease and population screening
De novo variants in MED12 cause X-linked syndromic neurodevelopmental disorders in 18 females
An autosomal dominant neurological disorder caused by de novo variants in FAR1 resulting in uncontrolled synthesis of ether lipids
Neurofilament light chain levels correlate with clinical measures in CLN3 disease
Toward newborn screening of metachromatic leukodystrophy: results from analysis of over 27,000 newborn dried blood spots
Evaluating the resource implications of different service delivery models for offering additional genomic findings
Impact of prenatal exome sequencing for fetal genetic diagnosis on maternal psychological outcomes and decisional conflict in a prospective cohort
Correction: Assessment of plasma lyso-Gb3 for clinical monitoring of treatment response in migalastat-treated patients with Fabry disease
High-throughput fetal fraction amplification increases analytical performance of noninvasive prenatal screening
Biallelic variants in HPDL, encoding 4-hydroxyphenylpyruvate dioxygenase-like protein, lead to an infantile neurodegenerative condition
Cardiovascular risk factors and body composition in adults with achondroplasia
Defining the genotypic and phenotypic spectrum of X-linked MSL3-related disorder
Biallelic inheritance of hypomorphic PKD1 variants is highly prevalent in very early onset polycystic kidney disease
Correction: A randomized controlled trial of an online health tool about Down syndrome
Correspondence on “The role of clinical response to treatment in determining pathogenicity of genomic variants” by Shen et al.
Evaluating variants classified as pathogenic in ClinVar in the DDD Study
New insights into the clinical and molecular spectrum of the novel CYFIP2-related neurodevelopmental disorder and impairment of the WRC-mediated actin dynamics
Impact of integrated translational research on clinical exome sequencing
NEXMIF encephalopathy: an X-linked disorder with male and female phenotypic patterns
Analysis of laboratory reporting practices using a quality assessment of a virtual patient
Exome sequencing analysis on products of conception: a cohort study to evaluate clinical utility and genetic etiology for pregnancy loss
Screening embryos for polygenic conditions and traits: ethical considerations for an emerging technology
The broad phenotypic spectrum of PPP2R1A-related neurodevelopmental disorders correlates with the degree of biochemical dysfunction
Correction: Long-awaited progress in addressing genetic discrimination in the United States
Cost-effectiveness of genome-wide sequencing for unexplained developmental disabilities and multiple congenital anomalies
Individuals with common diseases but with a low polygenic risk score could be prioritized for rare variant screening
Heterozygous variants that disturb the transcriptional repressor activity of FOXP4 cause a developmental disorder with speech/language delays and multiple congenital abnormalities
Dopachrome tautomerase variants in patients with oculocutaneous albinism
Clinical sites of the Undiagnosed Diseases Network: unique contributions to genomic medicine and science
The role of clinical response to treatment in determining pathogenicity of genomic variants
Correspondence on “Aminoacyl-tRNA synthetase deficiencies in search of common themes” by Fuchs et al.
Integration of functional assay data results provides strong evidence for classification of hundreds of BRCA1 variants of uncertain significance
Response to Shen and Zou
Geleophysic and acromicric dysplasias: natural history, genotype–phenotype correlations, and management guidelines from 38 cases
Cross-sectional quantitative analysis of the natural history of TUBA1A and TUBB2B tubulinopathies
A systematic review of monogenic etiologies of nonimmune hydrops fetalis
Correction: Genomic and sequence variants of protein kinase A regulatory subunit type 1β (PRKAR1B) in patients with adrenocortical disease and Cushing syndrome
Ceramide synthase TLCD3B is a novel gene associated with human recessive retinal dystrophy
JARID2 haploinsufficiency is associated with a clinically distinct neurodevelopmental syndrome
A phase 2/3 randomized clinical trial followed by an open-label extension to evaluate the effectiveness of elamipretide in Barth syndrome, a genetic disorder of mitochondrial cardiolipin metabolism
Genome sequencing increases diagnostic yield in clinically diagnosed Alagille syndrome patients with previously negative test results
Long-awaited progress in addressing genetic discrimination in the United States
Laboratory analysis of acylcarnitines, 2020 update: a technical standard of the American College of Medical Genetics and Genomics (ACMG)
Individuals with Down syndrome hospitalized with COVID-19 have more severe disease
Addendum: Technical standards and guidelines for spinal muscular atrophy testing
Disease-specific variant pathogenicity prediction significantly improves variant interpretation in inherited cardiac conditions
A model-based cost-effectiveness analysis of pharmacogenomic panel testing in cardiovascular disease management: preemptive, reactive, or none?
Heterozygous lamin B1 and lamin B2 variants cause primary microcephaly and define a novel laminopathy
Return of results in a global survey of psychiatric genetics researchers: practices, attitudes, and knowledge
CMAP changes upon symptom onset and during treatment in spinal muscular atrophy patients: lessons learned from newborn screening
Heterozygous truncating variants in SUFU cause congenital ocular motor apraxia
Correction: Initial experience from a renal genetics clinic demonstrates a distinct role in patient management
Response to Briuglia et al.
Ethical and public health implications of genetic testing for suicide risk: family and survivor perspectives
Correspondence on “Pathogenic variants in USP7 cause a neurodevelopmental disorder with speech delays, altered behavior, and neurologic anomalies” by Fountain et al.
Prospective clinical investigations of children with periodontal Ehlers–Danlos syndrome identify generalized lack of attached gingiva as a pathognomonic feature
Prospective phenotyping of long-term survivors of generalized arterial calcification of infancy (GACI)
Assessment of plasma lyso-Gb3 for clinical monitoring of treatment response in migalastat-treated patients with Fabry disease
Response to Thibodeau and Langlois
Correspondence on “The prevalence of genetic diagnoses in fetuses with severe congenital heart defects” by Nisselrooij et al.
Identifying rare, medically relevant variation via population-based genomic screening in Alabama: opportunities and pitfalls
A genotype-first approach to exploring Mendelian cardiovascular traits with clear external manifestations
(Un)standardized testing: the diagnostic odyssey of children with rare genetic disorders in Alberta, Canada
Impact of newborn screening and quality of therapy on the neurological outcome in glutaric aciduria type 1: a meta-analysis
Characterization of clinically relevant copy-number variants from exomes of patients with inherited heart disease and unexplained sudden cardiac death
Interpreting the impact of noncoding structural variation in neurodevelopmental disorders
Clinical impact of genomic testing in patients with suspected monogenic kidney disease
Correction: Aminoacyl-tRNA synthetase deficiencies in search of common themes
The NHGRI Short Course in Genomics: energizing genetics and genomics education in classrooms through direct engagement between educators and scientists
Correction: DOORS syndrome and a recurrent truncating ATP6V1B2 variant
Clinical and psychological outcomes of receiving a variant of uncertain significance from multigene panel testing or genomic sequencing: a systematic review and meta-analysis
Genomic and sequence variants of protein kinase A regulatory subunit type 1β (PRKAR1B) in patients with adrenocortical disease and Cushing syndrome
Predicting the risk of cardiac myxoma in Carney complex
Enhancing rare variant interpretation in inherited arrhythmias through quantitative analysis of consortium disease cohorts and population controls
Response to Faulkner et al.
Reassessment of causality of ABCC6 missense variants associated with pseudoxanthoma elasticum based on Sherloc
Correspondence on “Is there a duty to reinterpret genetic data? The ethical dimensions” by Appelbaum 17:39et al.
DOORS syndrome and a recurrent truncating ATP6V1B2 variant
Evidence in the UK Biobank for the underdiagnosis of erythropoietic protoporphyria
A novel statistical method for interpreting the pathogenicity of rare variants
Cell-free DNA as a diagnostic analyte for molecular diagnosis of vascular malformations
A randomized controlled trial of an online health tool about Down syndrome
Pathogenic variants in THSD4, encoding the ADAMTS-like 6 protein, predispose to inherited thoracic aortic aneurysm
The value of genomic sequencing in complex pediatric neurological disorders: a discrete choice experiment
Germline variants in HEY2 functional domains lead to congenital heart defects and thoracic aortic aneurysms
Lack of evidence to support recommendation for prenatal uniparental disomy (UPD) analysis following mosaic embryo transfer
Response to Mounts and Besser
Galactokinase deficiency: lessons from the GalNet registry
When phenotype does not match genotype: importance of “real-time” refining of phenotypic information for exome data interpretation
Assessment of technical heterogeneity among diagnostic tests to detect germline risk variants for hematopoietic malignancies
Addendum: American College of Medical Genetics and Genomics Standards and Guidelines for Clinical Genetics Laboratories, 2014 edition: technical standards and guidelines for Huntington disease
Ethical, social, and cultural issues related to clinical genetic testing and counseling in low- and middle-income countries: a systematic review
In This Issue
News
In This Issue
News
Correction to: An autosomal dominant neurological disorder caused by de novo variants in FAR1 resulting in uncontrolled synthesis of ether lipids
Correction to: Early cancer diagnoses through BRCA1/2 screening of unselected adult biobank participants
News
In This Issue
News
In This Issue
Correction to: Rare variants in KDR, encoding VEGF Receptor 2, are associated with tetralogy of Fallot
Correction: Phenotypic expansion of CACNA1C-associated disorders to include isolated neurological manifestations
Correction to: Screening for autosomal recessive and X-linked conditions during pregnancy and preconception: a practice resource of the American College of Medical Genetics and Genomics (ACMG)
Expanded phenotype of AARS1-related white matter disease
Correction: The persistent lack of knowledge and misunderstanding of the Genetic Information Nondiscrimination Act (GINA) more than a decade after passage
A systematic literature review of disclosure practices and reported outcomes for medically actionable genomic secondary findings
Evaluating the performance of a clinical genome sequencing program for diagnosis of rare genetic disease, seen through the lens of craniosynostosis
A qualitative investigation of biomedical informatics interoperability standards for genetic test reporting: benefits, challenges, and motivations from the testing laboratory’s perspective
The design, implementation, and effectiveness of intervention strategies aimed at improving genetic referral practices: a systematic review of the literature
Correction to: Focused Revision: Policy statement on folic acid and neural tube defects
Correction to: The genetic architecture of Plakophilin 2 cardiomyopathy
The persistent lack of knowledge and misunderstanding of the Genetic Information Nondiscrimination Act (GINA) more than a decade after passage
PPA2-associated sudden cardiac death: extending the clinical and allelic spectrum in 20 new families
Correction to: Cystic fibrosis–related diabetes onset can be predicted using biomarkers measured at birth
Positive predictive value highlights four novel candidates for actionable genetic screening from analysis of 220,000 clinicogenomic records
Safety and efficacy of low-dose PI3K inhibitor taselisib in adult patients with CLOVES and Klippel–Trenaunay syndrome (KTS): the TOTEM trial, a phase 1/2 multicenter, open-label, single-arm study
Impact of personal genomic risk information on melanoma prevention behaviors and psychological outcomes: a randomized controlled trial
Biallelic loss-of-function variants in the splicing regulator NSRP1 cause a severe neurodevelopmental disorder with spastic cerebral palsy and epilepsy
Genetic testing in ambulatory cardiology clinics reveals high rate of findings with clinical management implications
News
Delineating the molecular and phenotypic spectrum of the SETD1B-related syndrome
Correction to: ACMG SF v3.0 list for reporting of secondary findings in clinical exome and genome sequencing: a policy statement of the American College of Medical Genetics and Genomics (ACMG)
Integrative approach to interpret DYRK1A variants, leading to a frequent neurodevelopmental disorder
Breast cancer polygenic risk scores: a 12-month prospective study of patient reported outcomes and risk management behavior
Pathogenic variants in GNPTAB and GNPTG encoding distinct subunits of GlcNAc-1-phosphotransferase differentially impact bone resorption in patients with mucolipidosis type II and III
Safe and persistent growth-promoting effects of vosoritide in children with achondroplasia: 2-year results from an open-label, phase 3 extension study
In This Issue
Exploring the motivations of research participants who chose not to learn medically actionable secondary genetic findings about themselves
Large scale clinical exome sequencing uncovers the scope and severity of skin disorders associated with MC1R genetic variants
Tasimelteon safely and effectively improves sleep in Smith–Magenis syndrome: a double-blind randomized trial followed by an open-label extension
Residual risk for additional recessive diseases in consanguineous couples
Correction to: Direct-to-consumer prenatal testing for multigenic or polygenic disorders: a position statement of the American College of Medical Genetics and Genomics (ACMG)
Correction to: Neutropenia and intellectual disability are hallmarks of biallelic and de novo CLPB deficiency
Correction to: The underestimated burden of monogenic kidney disease in adults waitlisted for kidney transplantation
Screening for autosomal recessive and X-linked conditions during pregnancy and preconception: a practice resource of the American College of Medical Genetics and Genomics (ACMG)
Clinical and genomic characterization of 8p cytogenomic disorders
Opportunities and pitfalls of social media research in rare genetic diseases: a systematic review
Multisite investigation of strategies for the clinical implementation of pre-emptive pharmacogenetic testing
Caffey disease is associated with distinct arginine to cysteine substitutions in the proα1(I) chain of type I procollagen
Germline ATM variants predispose to melanoma: a joint analysis across the GenoMEL and MelaNostrum consortia
Safety and efficacy of N-acetylmannosamine (ManNAc) in patients with GNE myopathy: an open-label phase 2 study
Neptune: an environment for the delivery of genomic medicine
Increased risk for dementia in neurofibromatosis type 1
Correction to: Clinical spectrum of MTOR-related hypomelanosis of Ito with neurodevelopmental abnormalities
Challenges in genetic testing: clinician variant interpretation processes and the impact on clinical care
Uptake and acceptability of a mainstreaming model of hereditary cancer multigene panel testing among patients with ovarian, pancreatic, and prostate cancer
Pathogenic variants of meiotic double strand break (DSB) formation genes PRDM9 and ANKRD31 in premature ovarian insufficiency
Biallelic variants in PCDHGC4 cause a novel neurodevelopmental syndrome with progressive microcephaly, seizures, and joint anomalies
High rate of hypomorphic variants as the cause of inherited ataxia and related diseases: study of a cohort of 366 families
Disclosure of genetic information to family members: a systematic review of normative documents
Response to Resta et al.
Correction to: Cardiovascular risk factors and body composition in adults with achondroplasia
Ethical challenges for a new generation of early-phase pediatric gene therapy trials
Correspondence on “Clinical spectrum of MTOR-related hypomelanosis of Ito with neurodevelopmental abnormalities,” by Carmignac et al.
A qualitative study of prevalent laboratory information systems and data communication patterns for genetic test reporting
Clinical likelihood ratios and balanced accuracy for 44 in silico tools against multiple large-scale functional assays of cancer susceptibility genes
One is the loneliest number: genotypic matchmaking using the electronic health record
Potential of polygenic risk scores for improving population estimates of women’s breast cancer genetic risks
Focused Revision: Policy statement on folic acid and neural tube defects
Missense NAA20 variantsimpairing the NatB protein N-terminal acetyltransferase cause autosomal recessivedevelopmental delay, intellectual disability, and microcephaly
InpherNet accelerates monogenic disease diagnosis using patients’ candidate genes’ neighbors
Disease-specific ACMG/AMP guidelines improve sequence variant interpretation for hearing loss
Exome and genome sequencing for pediatric patients with congenital anomalies or intellectual disability: an evidence-based clinical guideline of the American College of Medical Genetics and Genomics (
Upregulation of GBP1 in thyroid primordium is required for developmental thyroid morphogenesis
Treatment of ARS deficiencies with specific amino acids
Clinical impact of re-evaluating genes and variants implicated in dilated cardiomyopathy
An integrative multiomics analysis identifies putative causal genes for COVID-19 severity
Direct-to-consumer prenatal testing for multigenic or polygenic disorders: a position statement of the American College of Medical Genetics and Genomics (ACMG)
Can you hear us now? The impact of health-care utilization by rare disease patients in the United States
News
Biallelic variants in KARS1 are associated with neurodevelopmental disorders and hearing loss recapitulated by the knockout zebrafish
Mucolipidosis type II and type III: a systematic review of 843 published cases
In This Issue
Phenotypic expansion of CACNA1C-associated disorders to include isolated neurological manifestations
Inherited duplications of PPP2R3B predispose to nevi and melanoma via a C21orf91-driven proliferative phenotype
Neutropenia and intellectual disability are hallmarks of biallelic and de novo CLPB deficiency
Genetic testing for inherited colorectal cancer and polyposis, 2021 revision: a technical standard of the American College of Medical Genetics and Genomics (ACMG)
Response to Suay-Corredera et al.
Response to Stern et al.
Genome-wide cell-free DNA screening: a focus on copy-number variants
Correspondence on “Prospective phenotyping of long-term survivors of generalized arterial calcification of infancy (GACI)” by Ferreira et al.
The genetic architecture of Plakophilin 2 cardiomyopathy
Chromosomal microarray analysis, including constitutional and neoplastic disease applications, 2021 revision: a technical standard of the American College of Medical Genetics and Genomics (ACMG)
Couple-based expanded carrier screening provided by general practitioners to couples in the Dutch general population: psychological outcomes and reproductive intentions
Truncating variants in the SHANK1 gene are associated with a spectrum of neurodevelopmental disorders
A framework for automated gene selection in genomic applications
Correspondence on “Computational prediction of protein subdomain stability in MYBPC3 enables clinical risk stratification in hypertrophic cardiomyopathy and enhances variant interpretation” by Thompso
Rare variants in KDR, encoding VEGF Receptor 2, are associated with tetralogy of Fallot
Haploinsufficiency of ARFGEF1 is associated with developmental delay, intellectual disability, and epilepsy with variable expressivity
Cost or price of sequencing? Implications for economic evaluations in genomic medicine
The predictive ability of the 313 variant–based polygenic risk score for contralateral breast cancer risk prediction in women of European ancestry with a heterozygous BRCA1 or BRCA2 pathogenic variant women of European ancestry
PIGG variant pathogenicity assessment reveals characteristic features within 19 families
Heterozygous loss-of-function variants significantly expand the phenotypes associated with loss of GDF11
GenomeDiver: a platform for phenotype-guided medical genomic diagnosis
Primary care providers’ responses to unsolicited Lynch syndrome secondary findings of varying clinical significance
Ask me later: deciding to have clinical exome trio sequencing for my critically ill child
Clinical utility of testing for PALB2 and CHEK2 c.1100delC in breast and ovarian cancer
Revised diagnostic criteria for neurofibromatosis type 1 and Legius syndrome: an international consensus recommendation
Biallelic and monoallelic variants in PLXNA1 are implicated in a novel neurodevelopmental disorder with variable cerebral and eye anomalies
Correspondence on “De novo variants in MED12 cause X-linked syndromic neurodevelopmental disorders in 18 females” by Polla et al.
Response to Riccardi et al.
X-linked creatine transporter deficiency results in prolonged QTc and increased sudden death risk in humans and disease model
News
In This Issue
Diagnosing newborns with suspected mitochondrial disorders: an economic evaluation comparing early exome sequencing to current typical care
SNORD116 and growth hormone therapy impact IGFBP7 in Prader–Willi syndrome
UBA2 variants underlie a recognizable syndrome with variable aplasia cutis congenita and ectrodactyly
Improved detection of mitochondrial DNA instability in mitochondrial genome maintenance disorders
Correction: Janus-faced EPHB4-associated disorders: novel pathogenic variants and unreported intrafamilial overlapping phenotypes
Quantitative analysis of the natural history of prolidase deficiency: description of 17 families and systematic review of published cases
Somatic PIK3R1 variation as a cause of vascular malformations and overgrowth
A randomized controlled trial of genetic testing and cascade screening in familial hypercholesterolemia
ACMG SF v3.0 list for reporting of secondary findings in clinical exome and genome sequencing: a policy statement of the American College of Medical Genetics and Genomics (ACMG)
Recommendations for reporting of secondary findings in clinical exome and genome sequencing, 2021 update: a policy statement of the American College of Medical Genetics and Genomics (ACMG)
Privacy practices using genetic data from cell-free DNA aneuploidy screening
Achondroplasia Natural History Study (CLARITY): a multicenter retrospective cohort study of achondroplasia in the United States
One in seven pathogenic variants can be challenging to detect by NGS: an analysis of 450,000 patients with implications for clinical sensitivity and genetic test implementation
Recontacting registry participants with genetic updates through GenomeConnect, the ClinGen patient registry
Severity modeling of propionic acidemia using clinical and laboratory biomarkers
Randomized prospective evaluation of genome sequencing versus standard-of-care as a first molecular diagnostic test
Management of individuals with germline variants in PALB2: a clinical practice resource of the American College of Medical Genetics and Genomics (ACMG)
Correction to: 1-13C-propionate breath testing as a surrogate endpoint to assess efficacy of liver-directed therapies in methylmalonic acidemia (MMA)
Genetic counseling and testing for Asian Americans: a systematic review
Genomic medicine implementation protocols in the PhenX Toolkit: tools for standardized data collection
Correction to: Disrupted intraflagellar transport due to IFT74 variants causes Joubert syndrome
Reproductive outcomes in individuals with chromosomal reciprocal translocations
Paired-like homeobox gene (PHOX2B) nonpolyalanine repeat expansion mutations (NPARMs): genotype–phenotype correlation in congenital central hypoventilation syndrome (CCHS)
Laboratory business models and practices: implications for availability and access to germline genetic testing
Correction to: Three years of clinical experience with a genome-wide cfDNA screening test for aneuploidies and copy number variants
Correction: A randomized trial to study the comparative efficacy of phenylbutyrate and benzoate on nitrogen excretion and ureagenesis in healthy volunteers
Erythropoietic protoporphyria: time to prodrome, the warning signal to exit sun exposure without pain—a patient-reported outcome efficacy measure
The 2019 US medical genetics workforce: a focus on clinical genetics
Loss-of-function and missense variants in NSD2 cause decreased methylation activity and are associated with a distinct developmental phenotype
Correspondence on “DOORS syndrome and a recurrent truncating ATP6V1B2 variant” by Beauregard-Lacroix et al.
Correction to: Pitfalls of clinical exome and gene panel testing: alternative transcripts
Response to Gao et al.
The genetic landscape of intellectual disability and epilepsy in adults and the elderly: a systematic genetic work-up of 150 individuals
Approaches to assessing the provider experience with clinical pharmacogenomic information: a scoping review
Retrospective analysis and reclassification of DYSF variants in a large French series of dysferlinopathy patients
Next-generation sequencing for constitutional variants in the clinical laboratory, 2021 revision: a technical standard of the American College of Medical Genetics and Genomics (ACMG)
Refining the risk for fragile X–associated primary ovarian insufficiency (FXPOI) by FMR1 CGG repeat size
Correction: The illusion of polygenic disease risk prediction
Correction to: Correspondence on “AminoacyltRNA synthetase deficiencies in search of common themes” by Fuchs et al.
Typical 22q11.2 deletion syndrome appears to confer a reduced risk of schwannoma
Combining exome/genome sequencing with data repository analysis reveals novel gene–disease associations for a wide range of genetic disorders
In This Issue
News
One-year results of a clinical trial of olipudase alfa enzyme replacement therapy in pediatric patients with acid sphingomyelinase deficiency
Incidental detection of acquired variants in germline genetic and genomic testing: a points to consider statement of the American College of Medical Genetics and Genomics (ACMG)
Janus-faced EPHB4-associated disorders: novel pathogenic variants and unreported intrafamilial overlapping phenotypes
Systematic analysis of short tandem repeats in 38,095 exomes provides an additional diagnostic yield
Interference of nuclear mitochondrial DNA segments in mitochondrial DNA testing resembles biparental transmission of mitochondrial DNA in humans
Variants in PRKAR1B cause a neurodevelopmental disorder with autism spectrum disorder, apraxia, and insensitivity to pain
Clinical spectrum of MTOR-related hypomelanosis of Ito with neurodevelopmental abnormalities
Haploinsufficiency of PRR12 causes a spectrum of neurodevelopmental, eye, and multisystem abnormalities
Videoconferencing to deliver genetics services: a systematic review of telegenetics in light of the COVID-19 pandemic
Biallelic variants in TMEM222 cause a new autosomal recessive neurodevelopmental disorder
Ensuring best practice in genomics education and evaluation: reporting item standards for education and its evaluation in genomics (RISE2 Genomics)
Penetrance and outcomes at 1-year following return of actionable variants identified by genome sequencing
Patients’ and professionals’ perspective of non-in-person visits in hereditary cancer: predictors and impact of the COVID-19 pandemic
1-13C-propionate breath testing as a surrogate endpoint to assess efficacy of liver-directed therapies in methylmalonic acidemia (MMA)
Laboratory testing for fragile X, 2021 revision: a technical standard of the American College of Medical Genetics and Genomics (ACMG)
From genes to public health: are we ready for DNA-based population screening?
DNA-based screening and personal health: a points to consider statement for individuals and health-care providers from the American College of Medical Genetics and Genomics (ACMG)
Establishing the value of genomics in medicine: the IGNITE Pragmatic Trials Network
Computational prediction of protein subdomain stability in MYBPC3 enables clinical risk stratification in hypertrophic cardiomyopathy and enhances variant interpretation
Utility of noninvasive genome-wide screening: a prospective cohort of obstetric patients undergoing diagnostic testing
Universal newborn genetic screening for pediatric cancer predisposition syndromes: model-based insights
Reducing Sanger confirmation testing through false positive prediction algorithms
Variant curation expert panel recommendations for RYR1 pathogenicity classifications in malignant hyperthermia susceptibility
In This Issue
Expanded clinical validation of Haploseek for comprehensive preimplantation genetic testing
Artificial intelligence–assisted phenotype discovery of fragile X syndrome in a population-based sample
Discordant results between conventional newborn screening and genomic sequencing in the BabySeq Project
Low-pass genome sequencing–based detection of absence of heterozygosity: validation in clinical cytogenetics
News
DNA-based screening and population health: a points to consider statement for programs and sponsoring organizations from the American College of Medical Genetics and Genomics (ACMG)
Response to Carlson
Correspondence on “Assessing relatives’ readiness for hereditary cancer cascade genetic testing” by Bednar et al.
Diagnostic exome-based preconception carrier testing in consanguineous couples: results from the first 100 couples in clinical practice
Correction: 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 (
Lysosomal cholesterol accumulation contributes to the movement phenotypes associated with NUS1 haploinsufficiency
Three years of clinical experience with a genome-wide cfDNA screening test for aneuploidies and copy-number variants
Computational analysis of 10,860 phenotypic annotations in individuals with SCN2A-related disorders
Clinical relevance of genotype–phenotype correlations beyond vascular events in a cohort study of 1500 Marfan syndrome patients with FBN1 pathogenic variants
The intersection of genetics and COVID-19 in 2021: preview of the 2021 Rodney Howell Symposium
The underestimated burden of monogenic kidney disease in adults waitlisted for kidney transplantation
Genome-wide investigation identifies a rare copy-number variant burden associated with human spina bifida
Correction to: De novo variants in SNAP25 cause an early-onset developmental and epileptic encephalopathy
High diagnosis rate for nonimmune hydrops fetalis with prenatal clinical exome from the Hydrops-Yielding Diagnostic Results of Prenatal Sequencing (HYDROPS) Study
Clinical delineation, sex differences, and genotype–phenotype correlation in pathogenic KDM6A variants causing X-linked Kabuki syndrome type 2
Addendum: American College of Medical Genetics consensus statement on factor V Leiden mutation testing
The role of digital tools in the delivery of genomic medicine: enhancing patient-centered care
Disruption of RFX family transcription factors causes autism, attention-deficit/hyperactivity disorder, intellectual disability, and dysregulated behavior
Taking an antiracist posture in scientific publications in human genetics and genomics
Correction: Clinical epigenomics: genome-wide DNA methylation analysis for the diagnosis of Mendelian disorders
The effect of early hormonal treatment (EHT) on expressive and receptive language capabilities in boys with 47,XXY (Klinefelter syndrome) during infancy and early childhood
Expanding evidence leads to new pharmacogenomics payer coverage
Response to Park et al.
News
In This Issue
Systematic assessment of outcomes following a genetic diagnosis identified through a large-scale research study into developmental disorders
DLG4-related synaptopathy: a new rare brain disorder
Preferences of biobank participants for receiving actionable genomic test results: results of a recontacting study
Trainee perspectives of COVID-19 impact on medical genetics education
Call for improvement in medical school training in genetics: results of a national survey
Commonalities across computational workflows for uncovering explanatory variants in undiagnosed cases
When to test fetuses for RASopathies? Proposition from a systematic analysis of 352 multicenter cases and a postnatal cohort
Clinical and laboratory reporting impact of ACMG-AMP and modified ClinGen variant classification frameworks in MYH7-related cardiomyopathy
Deep phenotyping in 3q29 deletion syndrome: recommendations for clinical care
Outcome of publicly funded nationwide first-tier noninvasive prenatal screening
Correspondence on “Clinical, neuropathological, and genetic characterization of STUB1 variants in cerebellar ataxias: a frequent cause of predominant cognitive impairment” by Roux et al.
Clinical epigenomics: genome-wide DNA methylation analysis for the diagnosis of Mendelian disorders
Evaluating the molecular diagnostic yield of joint genotyping–based approach for detecting rare germline pathogenic and putative loss-of-function variants
Disrupted intraflagellar transport due to IFT74 variants causes Joubert syndrome
GUÍA: a digital platform to facilitate result disclosure in genetic counseling
A recurrent, homozygous EMC10 frameshift variant is associated with a syndrome of developmental delay with variable seizures and dysmorphic features
Focused Revision: ACMG practice resource: Genetic evaluation of short stature
Deep phenotyping unstructured data mining in an extensive pediatric database to unravel a common KCNA2 variant in neurodevelopmental syndromes
Systematic large-scale assessment of the genetic architecture of left ventricular noncompaction reveals diverse etiologies
Development and implementation of an electronic medical record module to track genetic testing results
Cystic fibrosis–related diabetes onset can be predicted using biomarkers measured at birth
Increasing access to individualized medicine: a matched-cohort study examining Latino participant experiences of genomic screening
Deletion of the NKG2C receptor encoding KLRC2 gene and HLA-E variants are risk factors for severe COVID-19
Transforming the clinical outcome in CRIM-negative infantile Pompe disease identified via newborn screening: the benefits of early treatment with enzyme replacement therapy and immune tolerance induct
Loss-of-function variants in SEMA3F and PLXNA3 encoding semaphorin-3F and its receptor plexin-A3 respectively cause idiopathic hypogonadotropic hypogonadism
Impact of newborn screening on the reported incidence and clinical outcomes associated with medium- and long-chain fatty acid oxidation disorders
In This Issue
Unsuspected somatic mosaicism for FBN1 gene contributes to Marfan syndrome
CDK19-related disorder results from both loss-of-function and gain-of-function de novo missense variants
News
Uniparental disomy in a population of 32,067 clinical exome trios
Ethnicity has a multiplex impact upon the risk of a full mutation expansion among female heterozygotes for FMR1 premutation
Variants in GNAI1 cause a syndrome associated with variable features including developmental delay, seizures, and hypotonia
Outcomes of prior authorization requests for genetic testing in outpatient pediatric genetics clinics
A pathogenic UFSP2 variant in an autosomal recessive form of pediatric neurodevelopmental anomalies and epilepsy
The role of phenotype-based search approaches using public online databases in diagnostics of Mendelian disorders
Is precision medicine relevant in the age of COVID-19?
Prevalence of pathogenic germline variants in patients with metastatic renal cell carcinoma
Beyond diagnostic yield: prenatal exome sequencing results in maternal, neonatal, and familial clinical management changes
Newborn bloodspot screening in the time of COVID-19
Biallelic truncating variants in MAPKAPK5 cause a new developmental disorder involving neurological, cardiac, and facial anomalies combined with synpolydactyly
Phenotypic continuum between Waardenburg syndrome and idiopathic hypogonadotropic hypogonadism in humans with SOX10 variants
Methods and feasibility study for exome sequencing as a universal second-tier test in newborn screening
Private payer coverage policies for ApoE-e4 genetic testing
Patients dispensed medications with actionable pharmacogenomic biomarkers: rates and characteristics
A hybrid implementation-effectiveness randomized trial of CYP2D6-guided postoperative pain management
Response to Biesecker et al.
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