American Journal of Medical Genetics Part A - 2024

410 articles | Last updated: 2025-12-03 14:12:55
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T A T A T A T A
Genetic Insights Into Epidermolysis Bullosa: Identification of Novel Variants in Tunisian Patients North African; Northern Africa; Tunisian; Tunisia
A Homozygous <scp><i>MYH1</i></scp> Variant Underlies Autosomal Recessive Isolated Recurrent Rhabdomyolysis
Cerebrospinal Fluid and Serum Neuron‐Specific Enolase in Niemann‐Pick Disease Type <scp>C1</scp>
<scp><i>COL4A2</i></scp>‐Related Disorder Presenting in Adulthood With Rhabdomyolysis
Transcriptomic and Metabolomic Analyses in Monozygotic and Dizygotic Twins
Community‐Sourced Reporting of Mortalities in Angelman Syndrome (1979–2022)
In This Issue
Long‐Read Sequencing Increases Diagnostic Yield for Rare Disease
Table of Contents, Volume 197A, Number 1, January 2025
Repeat Expansion Disorders Likely Underdiagnosed
Proband‐Only Exome Sequencing for Intellectual Disability in Iran: Diagnostic Yield and Genetic Insights
Gonadal Mosaicism for an <i>ASH1L</i> Intragenic Deletion Makes a Bridge Between <scp>MRD52</scp> and 1q22 Microdeletion
A Homozygous Nonsense Variant in the <scp><i>BICC1</i></scp> Gene Associated With Fetal Cystic Kidney Disease and Lower Limb Post‐Axial Polydactyly
Vitamin <scp>B5</scp> Monotherapy Improves Symptoms in a 7‐Year‐Old Girl With <scp>TANGO2</scp> Deficiency Disorder Chinese
The Natural History of Dermatosparaxis Ehlers Danlos Syndrome: An Adult Case Series
<scp>CHIME</scp> Syndrome in a Child With Homozygous <scp><i>PIGL</i> p.Leu167Pro</scp> Variant
Does It Run in Your Family? Inherited Truncating <scp><i>PSMD12</i></scp> Variants Broaden the Phenotypic Spectrum of Stankiewicz‐Isidor Syndrome
Immune Dysregulation in a Child With <i>SOD1</i>‐Related Neurological Disease
Telehealth Is Effective in the Evaluation of Individuals With Undiagnosed Rare Disorders: An Undiagnosed Diseases Network Study
Exploring the Clinical Spectrum of <scp><i>HUWE1</i></scp>‐Related Neurodevelopmental Disorder: Five New Patients and Literature Review
Constitutional Mosaic Pericentromeric Trisomy 8 in a Female Patient With Aplastic Anemia
Alu‐Mediated Deletion of <scp><i>FANCA</i></scp> in Turkish Families With Fanconi Anemia: Evidence of a Founder Effect Turkish population; Turkish descent
Subacute Neuropathy Post‐Liver Transplantation in Zellweger Spectrum Disorder: A Case Report
<scp>SURF1</scp> Deficiency: Expanding on Disease Phenotype and Assessing Disease Burden by Describing Clinical and Biochemical Phenotype
Dr. Peter Emil Becker and the Third Reich: Correspondence racial biology and eugenics
From Desbuquois Dysplasia to Multiple Epiphyseal Dysplasia: The Clinical Impact of a <scp><i>CANT1</i></scp> Variant Across Five Unrelated Families
Implementing a Quality Improvement Initiative to Screen for Dementia in a Down Syndrome Specialty Clinic
2q13 Distal Microdeletion: Considering Evidence for an Emerging Syndrome Versus Susceptibility Locus: Twenty‐Five New Cases and Review of the Literature
Health Care Transition Programs for Adolescents and Young Adults With Hereditary Cancer Predisposition: A Scoping Review
An Extended Phenotype of <i>PPP1R13L</i> Cardiocutaneous Syndrome
<i>De Novo</i> Balanced Translocations Disrupting the <scp><i>FBN1</i></scp> Gene Diagnosed by Genome Sequencing: An Uncommon Cause of Marfan Syndrome Modifying Genetic Counseling
Quality of Life in Short Stature Children With Skeletal Dysplasia: A Cross Sectional Study Using the Quality of Life in Short Stature Youth Questionnaire
Chronic Catatonia in an Individual With a De Novo Missense <i>SHANK1</i> Variant
From Clinical Observation to Genetic Confirmation: Somatic Mosaic Mutations in <i>RHOA</i> on Ectodermal Dysplasia With Multi‐System Involvement
Long‐Term Health Outcomes of Individuals With Pseudodeficiency Alleles in <scp><i>IDUA</i></scp> May Inform Newborn Screening Practices for Mucopolysaccharidosis Type I
A Novel De Novo Splice Acceptor Variant in <i>BICD2</i> Is Associated With Spinal Muscular Atrophy
The Glu86 Residue in <scp>TBX4</scp> Proves Critical for Human Lung Development
Insights From a Novel Splicing Variant and Recurrent Arginine Variants in the <scp><i>CHD3</i></scp> Gene Causing Snijders Blok–Campeau Syndrome
Characterizing and Evaluating the Structures of Combined Pediatrics and Medical Genetics and Genomics Residency Programs
Optical Genome Mapping (<scp>OGM</scp>) Identifies Multiple Structural Variants in a Case With Atypical Phelan‐<scp>McDermid</scp> Syndrome
Coffin‐Siris Syndrome and Unusual Angiogenic Profiles in Pregnancy: A Case Study Emphasizing Caution in Interpreting a Very Low <scp>sFlt</scp>‐1/<scp>PlGF</scp> Ratio
A second <scp>RUBCN</scp> variant associated with epileptic encephalopathy and neurodevelopmental delay
Expanding the Genetic and Phenotypic Spectrum of Mowat‐Wilson Syndrome: A Study of 10 Turkish Patients With an Intrafamilial Recurrence Caused by First Intragenic Large Deletion
Hailey‐Hailey Disease Caused by a Novel Deep Intronic Variant in <scp><i>ATP2C1</i></scp>
Structural Variants in <scp><i>COL1A1</i></scp> and <scp><i>COL1A2</i></scp> in Osteogenesis Imperfecta Swedish
Ring Chromosome 17 Syndrome—A Case Report and Discussion of Diagnostic Methods
Ethics of DNA Testing to Determine Nationality
Table of Contents, Volume 194A, Number 12, December 2024
Expanding the Molecular and Clinical Phenotype of Patients With <i>De Novo</i> Variants in <i>KIF5C</i>: A Six Patient Case Series
Gene Therapy Delivery Vector Crosses the Blood‐Brain Barrier
Cover Image, Volume 194A, Number 12, December 2024
In This Issue
Delayed Diagnosis of Spinal Muscular Atrophy in Two Chinese Families due to Novel <i>SMN1</i> Deletions Chinese
Identification and Characterization of Novel <scp>FSHR</scp> Copy Number Variations Causing Premature Ovarian Insufficiency
<i>CTNND1</i>‐Related Disorder: New Insight on Prenatal Phenotype
Cyclic Vomiting Syndrome in Patients Affected by Jansen–de Vries Syndrome: Results From an International Survey
Successful Pregnancy Outcome With Preconception Care in a Symptomatic Carrier of Duchenne Muscular Dystrophy: Case Report and Literature Review
New Insights Into the Spectrum of <scp>RASopathies</scp>: Clinical and Genetic Data in a Cohort of 121 Spanish Patients
<i>ARSA</i> Variant Associated With Late Infantile Metachromatic Leukodystrophy and Carrier Rate in Individuals of Ashkenazi Jewish Ancestry Ashkenazi Jewish; phrases: 'Ashkenazi Jewish ancestry', 'Ashkenazi Jewish descent', 'Ashkenazi Jewis
Recurrent Increased Nuchal Translucency Led to the Identification of Novel <i>NUP107</i> Variants
Neuroimaging to Genotype: Delineating the Spectrum of Disorders With Deficient Myelination in the Indian Population Indian Population
Further Evidence for a Possible Role for <i>ZFHX4</i> in Human Ocular Development and Disease
<scp><i>SOS1</i></scp>‐Related Noonan Syndrome and Sudden Cardiac Arrest in the Absence of Cardiomyopathy—An Arrhythmia Phenotype?
Expanding the Phenotype of Extremely Early Onset Juvenile Huntington's Disease: A Case Report and Review of Previously Published Cases
Recurrent Xp22.31‐Yq11 Unbalanced Translocations: Molecular Diagnosis and Clinical Implications in Three Families
A Prevalent <scp><i>TMEM260</i></scp> Deletion Causes Conotruncal Heart Defects, Including Truncus Arteriosus Japanese population; Japanese patients; Japanese patients with TA
Second Report of the <scp>p.Leu874Pro</scp> Missense Variant in <scp><i>EPHB4</i></scp> in a Family With Capillary Malformation‐Arteriovenous Malformation Syndrome (<scp>CM</scp>‐<scp>AVM</scp>) Syndr
Clinical Features of a Japanese Girl With Radio‐Tartaglia Syndrome due to a <i>SPEN</i> Truncating Variant Japanese
Expanding the Phenotype of <scp><i>NRROS</i></scp>‐Related <scp>SENEBAC</scp> Syndrome
Exome Sequencing Detects Uniparental Disomy of Chromosome 4 Revealing a <scp><i>LARP7</i></scp> Pathogenic Variant Responsible for Alazami Syndrome: A Case Report
<i>KIF11</i> Variants Associated With Novel Renal System Involvement—Two Cases That Expand the Phenotypic Spectrum of Microcephaly With or Without Chorioretinopathy, Lymphedema, or Impaired Intellectu
Cerebellar Hypoplasia and Treatment Course of a Two‐Month‐Old Infant With <scp><i>KCNQ2</i></scp> Epileptic Encephalopathy Due to a De Novo Variant and Review of the Literature
<scp>KBG</scp> Syndrome in 16 Indian Individuals Indian
An Unusual Presentation of Leber Hereditary Optic Neuropathy‐Plus Case Caused by a Novel <i>DNAJC30</i> Variant
Genetic Factors Linked to Early Clinical Trial Termination
Potential Therapy Corrects Calcium Signaling in Timothy Syndrome
In This Issue
Table of Contents, Volume 194A, Number 11, November 2024
Clinical and Genetic Spectrum of Patients With Mitochondrial Disease in a Pediatric Egyptian Cohort: Novel Variants and Phenotypic Expansion
Advancing Equity in Rare Disease Research: Insights From the Undiagnosed Disease Network Hispanic, Black non‐Hispanic, White non‐Hispanic, Asian non‐Hispanic, historically marginalized ethn
Managing <scp><i>CDH1</i></scp> Cancer Risks in a Child: Complex Decision Making in a Family With Hereditary Diffuse Gastric Cancer
A Distinctive Type of Mosaic Variegated Aneuploidy: Case Report and Review of the Literature
Bullous Lung Disease in Turner Syndrome: An Underrecognized Comorbidity?
Non‐Hotspot <i>PIK3CA</i> Variants Have Higher Variant Allele Frequency and are More Common in Syndromic Vascular Malformations
Neurodevelopment in Young Children With Sex Chromosome Trisomies Diagnosed Before Birth: A Cluster Analysis Study
Evidence That Long‐Term Treatment Prevents Tissue Oxidative Damage in Patients With Inherited Disorders of the Propionate Pathway
Periodic Paralysis in a Child With Thermosensitive Mitochondrial Trifunctional Protein Deficiency
Promoter Deletion Leading to Allele Specific Expression in a Genetically Unsolved Case of Primary Ciliary Dyskinesia
Assessment of Adaptive Functioning and the Impact of Seizures in <scp>KBG</scp> Syndrome
Examining Roles, Challenges, and Opportunities Within the Metabolic Genetics Workforce
Prevalence of Individuals With Multiple Diagnosed Genetic Diseases in the Undiagnosed Diseases Network
Bi‐Allelic Splicing Variant, c.153‐<scp>2A</scp> &gt; C in <scp><i>TOMM7</i></scp> Is Associated With Leigh Syndrome
Severe Phenotype With <scp>RECQL4</scp> Syndrome: A Report of Two Cases
Case Report of Friedreich's Ataxia and <scp><i>ALG1</i></scp>‐Related Biochemical Abnormalities in a Patient With Progressive Spastic Paraplegia
Evaluating the Influence of Social Determinants of Health on Blood Phenylalanine Levels in Phenylketonuria Patients
<scp><i>SF3B2</i></scp> Haploinsufficiency Associated With Hirschprung Disease and Complex Cardiac Defect Without Craniofacial Microsomia
Associated Anomalies in Radial Ray Deficiency
Hospital Visits Associated With Oral Infections in Patients With Neurofibromatosis Type 1: A Register‐Based Analysis
Craniotubular Dysplasia Ikegawa Type: Further Delineation of the Phenotype Indian
Seven Novel Variants of Weiss‐Kruszka Syndrome and Phenotype Expansion
Assessing Postnatal Mortality in Smith–Lemli–Opitz Syndrome
First Trimester Fetal Clubfoot: A Novel Presentation of Severe Osteogenesis Imperfecta
Germline <scp><i>RTEL1</i></scp> Variants in Telomere Biology Disorders
X‐Linked Bilateral Polymicrogyria With Epilepsy and Intellectual Disability Associated With a Novel <scp><i>KIF4A</i></scp> Variant
Diagnostic Accuracy of a Custom Large Language Model on Rare Pediatric Disease Case Reports
Whole‐Genome Sequencing Can Improve Care in Pediatric Cancer
A New Ocular Phenotype Combining Juvenile Glaucoma and Doyne Honeycomb Retinal Dystrophy (Malattia Leventinese) due to a Novel <i>EFEMP1</i> Pathogenic Variant
Study Strengthens Link between Autism Spectrum Disorder and Gut Microbiome
In This Issue
Cover Image, Volume 194A, Number 10, October 2024
Table of Contents, Volume 194A, Number 10, October 2024
Novel Clinical Manifestation and Favorable Treatment Outcome of Cochlear Implant in a Chinese Family With Likely Pathogenic Variant of the <scp><i>P2RX2</i></scp> Gene
Further Delineation of the Proximal 16p11.2 Microdeletion Syndrome: Novel Findings Among 22 New Individuals
Let Us Care for the Rare
Aortic Root Dilation and Genotype Associations in Phelan‐<scp>McDermid</scp> Syndrome
Loss‐of‐Function Variant in <scp><i>PPP1R12A</i></scp>‐Related Urogenital and/or Brain Malformation Syndrome: Expanded Phenotype of Sex Reversal Mexican
A Comparison of Clinical and Radiological Presentations of Sporadic and Tuberous Sclerosis Complex‐Associated Lymphangioleiomyomatosis
Investigating <scp><i>TNNC1</i></scp> gene inheritance and clinical outcomes through a comprehensive familial study
<i>WDR44</i> Loss‐of‐Function Promoter Deletion in a Male Newborn With a Ciliopathy Phenotype
Two novel compound heterozygous <scp><i>HOXB1</i></scp> variants in congenital facial palsy: A case report and a brief review of the literature
Expanding <scp>MNS1</scp> Heterotaxy Phenotype
Genetic Contributions to Lower Urinary Tract Dysfunction
Bridging the Diagnostic Gap for Hypermobile Ehlers‐Danlos Syndrome and Hypermobility Spectrum Disorders: Evidence of a Common Extracellular Matrix Fragmentation Pattern in Patient Plasma as a Potentia
Bilateral Perisylvian Polymicrogyria, Intellectual Disability and Nephronophthisis Associated With Compound Heterozygous Pathogenic Variants in the <scp><i>CEP83</i></scp> Gene
<i>MED12</i> Loss‐of‐Function Variants as a Cause of Congenital Diaphragmatic Hernia in Females With Hardikar Syndrome and Nonspecific Intellectual Disability
<scp><i>PRKAG2</i></scp>‐Related Lethal Congenital Glycogen Storage Disease of the Heart as Rare Cause of Fetal Hydrops With Bradycardia and Cardiomyopathy: Clinical Report and Literature Review
Intrafamilial Phenotypic Variability in <scp>SYNE1</scp>‐Related Disorder
Adherence to adult clinical practice guidelines for Down syndrome
Deletions in the <i>CDKL5</i> 5′ untranslated region lead to CDKL5 deficiency disorder
Chromosomal translocation resolves a diagnostic odyssey for familial <scp>Ruvalcaba</scp> syndrome
Recessive loss‐of‐function variants in <scp><i>DPH1</i></scp> identified as the molecular cause in a sibling pair previously diagnosed with Fine–Lubinsky syndrome
Diverse clinical presentation of <i>SPTBN1</i> variants: Complex versus primary attention‐deficit/hyperactivity disorder
Aberrant behavior checklist in youth with Prader–Willi syndrome: Preliminary study of cross‐sectional and longitudinal behavior characterization
Further evidence for an attenuated phenotype of in‐frame <i>DMD</i> deletions affecting the central rod domain of dystrophin around exon 48
Phenotypic spectrum and tumor risk in Simpson‐Golabi‐Behmel syndrome: Case series and comprehensive literature review
Recurrent carotid paragangliomas in a syndromic patient with a heterozygous missense variant in <i>DNA Methyltransferase 3 Alpha</i> African American
Recurrent p.<scp>H119Y</scp> variant in <scp><i>MAP2K1</i></scp> expands the phenotypic spectrum of <scp><i>MAP2K1</i></scp>‐related RASopathy
<scp><i>CCDC47</i></scp> gene and trichohepatoneurodevelopmental syndrome: Report of the fifth and sixth cases from <scp>Saudi Arabia</scp> Saudi origin; from Saudi Arabia
Novel heterozygous <scp><i>OPA3</i></scp> variant in a family with congenital cataracts, sensorineural hearing loss and neuropathy, without optic atrophy and comparison of pathogenic and population va
Comprehensive exploration of <scp>FCHO1</scp> mutations: <scp>Clinical</scp> manifestations and implications across disorders
Double somatic mosaicism in Marfan syndrome
<scp><i>CNOT1</i></scp> p.<scp>Arg535Cys</scp> variant in holoprosencephaly with late onset diabetes mellitus
Longitudinal echocardiography in pediatric patients with hypermobile Ehlers‐Danlos syndrome
Atypical presentation of <scp>ACCES</scp> syndrome resembling dominant Spondyloepiphyseal dysplasia tarda
<scp>Regions of Homozygocity</scp> size patterns among diverse ethnic groups in Israel: Toward tailored diagnostic reporting thresholds
Heterozygous loss of function variants in <scp><i>IFT140</i></scp> are associated with polycystic kidney disease
Prevalence rates for ectodermal dysplasia syndromes
Improving genetic testing utilization in a tertiary care neonatal intensive care unit through quality improvement
Correction to “Uniparental disomy of multiple chromosomes in two cases with a complex phenotype”
In This Issue
Accessible Molecular Testing Needed in Low Resource Settings
Table of Contents, Volume 194A, Number 9, September 2024
Noncoding Gene Identified as Relatively Common Cause of Intellectual Disability
Missense variant in <scp><i>PIGM</i></scp> associated with severe cystic encephalomalacia and portal vein thrombosis: Phenotypic and genotypic expansion of the glycosylphosphatidylinositol biosynthesi
Atypical noncontiguous <i>TSC2/PKD1</i> gene deletions presenting as tuberous sclerosis/polycystic kidney disease contiguous gene syndrome
Presentation of ichthyosis after substrate reduction therapy in Gaucher type 1
A novel <i>de novo</i> synonymous variant in <scp><i>GREB1L</i></scp> impacts the <scp>mRNA</scp> splicing associated with aplasia of the urogenital system
Gastrointestinal manifestations in Williams syndrome: A prospective analysis of an adult and pediatric cohort
Examining the impact of Native American myopathy on the quality of life and healthcare accessibility of patients and caregivers Native American
Clinical, genetic, and neuroimaging profiles of autosomal recessive spinocerebellar ataxia type 4 caused by novel <scp><i>VPS13D</i></scp> variants in <scp>Chinese</scp>
Prenatal molecular diagnosis of pyruvate dehydrogenase complex deficiency enables rapid initiation of ketogenic diet
Long‐read sequencing and optical genome mapping identify causative gene disruptions in noncoding sequence in two patients with neurologic disease and known chromosome abnormalities
Nablus mask‐like facial syndrome: Report of an atypical case with 8q21.3–q22.1 deletion
Novel causative variants in Legius syndrome: <i>SPRED1</i> Genotype spectrum expansion
Utility of genome sequencing in exome‐negative pediatric patients with neurodevelopmental phenotypes
A novel <i>ABCC9</i> variant in a <scp>Greek</scp> family with <scp>Cantu</scp> syndrome affecting multiple generations highlights the functional role of the <scp>SUR2B NBD1</scp> Greek
Subtle phenotypes of Mowat–Wilson syndrome in a patient with a novel <scp><i>ZEB2</i></scp> C‐<scp>ZF</scp> domain variant
Optical genome mapping with genome sequencing identifies subtelomeric <scp>Xq28</scp> deletion and inserted 7p22.3 duplication in a male with multisystem developmental disorder
Ophthalmic manifestations of <scp><i>NAA10</i></scp>‐related and <scp><i>NAA15</i></scp>‐related neurodevelopmental syndromes: Analysis of cortical visual impairment and refractive errors
Prevalence, diagnostic features, and medical outcomes of females with Turner syndrome with a trisomy X cell line (45,X/47,<scp>XXX</scp>): Results from the <scp>InsighTS</scp> Registry
Expanded phenotypic spectrum of <scp>UDP</scp>‐glucose‐6‐dehydrogenase recessive neurodevelopmental disorder: Two novel descriptions with or without epileptic encephalopathy
Expanding the clinical phenotype and variant spectrum associated with <scp><i>RFX7</i></scp> Afro‐Caribbean
Expansion of the phenotypic spectrum of <i>KARS1</i>‐related disorders to include arthrogryposis multiplex congenita and summary of experiences with lysine supplementation
Occurrence of cancer in Marfan syndrome: Report of two patients with neuroblastoma and review of the literature
Unveiling hidden genetic complexity: Coexistence of <scp><i>HGSNAT</i></scp> and <scp><i>EYS</i></scp> variants in a patient with retinal dystrophy
Are <i>SCN2A</i>‐related BFNISs also responsible for seizures in adulthood? A case report opens new scenarios
<i>APOE4</i> Homozygotes Represent a Distinct Genetic Subtype of Alzheimer's Disease
In This Issue
<i>EMILIN1</i> gene variant associated with polyneuropathy, language impairment, and motor dysfunction
Rule Expansion Further Regulates Laboratory Developed Tests
Table of Contents, Volume 194A, Number 8, August 2024
Erratum to “Cat eye syndrome: Clinical, cytogenetics and familial findings in a large cohort of 43 patients highlighting the importance of congenital heart disease and inherited cases”
Nerve enlargement in patients with Noonan syndrome: A retrospective cohort study
Molecular autopsy in Chinese sudden cardiac death in the young
First copy number variant <i>in trans</i> with single nucleotide variant in <i>CCN6</i> causing progressive pseudorheumatoid dysplasia revealed by genome sequencing and deep phenotyping in monozygotic
Medical students' reproductive health perspectives: Pre‐ and post‐<i>Roe v Wade</i> reversal
<scp><i>COL4A1</i></scp>‐related disorder as a mimic of congenital <scp>TORCHES</scp> infection—Expanding the clinical, neuroimaging and genotype spectrum
A unique case of hyperammonemia due to <scp>CA5A</scp> deficiency: Impact of coexisting gene mutations, pseudogene, and microdeletion
Secondary findings in genes related to cancer phenotypes in Turkish exome sequencing data from 2020 individuals Turkish population
Leukoencephalopathy with calcifications, developmental brain abnormalities and skeletal dysplasia due to homozygosity for a hypomorphic <scp><i>CSF1R</i></scp> variant: A report of three siblings
Another face of <i>RASA1</i>: Report of familial germline variant in <i>RASA1</i> with dysmorphic features
Joint contractures is a recurrent clinical feature of individuals with neurodevelopmental disorder due to <scp><i>FOXP1</i></scp> likely gene disruptive variants
<scp><i>ACOX1</i></scp> gain‐of‐function variation in a 10‐years‐old patient responsive to immunomodulating therapy
Genome sequencing identifies biallelic variants in <i>SCLT1</i> in a patient with syndromic nephronophthisis: Reflections on the <i>SCLT1</i>‐related ciliopathy spectrum
Low‐pass whole genome sequencing as a cost‐effective alternative to chromosomal microarray analysis for low‐ and middle‐income countries
Sleep correlates of behavior functioning in Cornelia de Lange syndrome
The motivations and methods behind sharing a pediatric <scp>Prader–Willi</scp> syndrome diagnosis
Reclassification of an <i>FBN1</i> variant emphasizes the importance of segregation analysis, information sharing, and multidisciplinary teamwork in understanding genetic variants in health and diseas
A novel missense variant in <i>HIKESHI</i>: Clinical phenotype, in vitro functional testing, and potential for gene therapy
Two siblings with acute necrotizing encephalopathy associated with variants of <scp><i>LARS1</i></scp>
Long‐term survival of an infant with complete tetraploidy: A case report
Reanalysis of <scp>RNA</scp> sequencing data ends diagnostic odyssey and expands the phenotypic spectrum of congenital titinopathy
<scp><i>GPC4</i></scp> truncating variant associated with Keipert syndrome and lacrimal punctal agenesis
The diagnostic yield of genetic and metabolic investigations in syndromic and nonsyndromic patients with autism spectrum disorder, global developmental delay, or intellectual disability from a dedicat
Known pathogenic gene variants and new candidates detected in sudden unexpected infant death using whole genome sequencing
Expanding the phenotype of <scp>Harel‐Yoon</scp> syndrome: A case report suggesting a genotype/phenotype correlation
Impact of definitive surgery for esophageal atresia on long‐term outcomes in patients with trisomy 18
New insights into the landscape of <i>ALPL</i> gene variants in patients with hypophosphatasia from the Global <scp>HPP</scp> Registry
Further defining the molecular spectrum and long‐term follow‐up of 17 patients with Dyggve–Melchior–Clausen and Smith–<scp>McCort</scp> dysplasia type 2
Germline <scp><i>PTCH1</i></scp>: <scp>c.361_362insAlu</scp> alteration identified by comprehensive exome and <scp>RNA</scp> sequencing in a patient with <scp>Gorlin</scp> syndrome
Pathogenic <scp><i>FLNA</i></scp> variants affecting the hinge region of filamin A are associated with male survival
A founder deletion in <scp><i>ECM1</i></scp> of 1163 bp causes lipoid proteinosis in the southeast region of Turkiye
A novel homozygous pathogenic missense variant in <scp><i>COX6B1</i></scp>: Further delineation of the phenotype
Exploring the Low Uptake of Gene Therapy in Hemophilia
Cover Image, Volume 194A, Number 7, July 2024
In This Issue
<i>TEK</i> gene‐related primary congenital glaucoma: Phenotypic features and mutational spectrum in a <scp>Mexican</scp> cohort of 10 unrelated families Mexican
Noonan syndrome and type 1 Chiari malformation: Possible association
Table of Contents, Volume 194A, Number 7, July 2024
New Gene Therapy Changes Treatment Landscape for Metachromatic Leukodystrophy
Relationship of thyroid function with genetic subtypes and treatment with growth hormone in <scp>Prader–Willi</scp> syndrome
<scp>NANS</scp>‐<scp>CDG</scp>: Expanding clinical insights with a novel patient with novel variants Korean
Expanded phenotype and cancer risk in patients with <scp>Beckwith–Wiedemann</scp> spectrum caused by <scp><i>CDKN1C</i></scp> variants
Siblings with vitamin D‐dependent rickets type <scp>1A</scp>: Importance of genetic testing and a review of genotype–phenotype correlations
Multicenter appraisal of comorbid <scp>TANGO2</scp> deficiency disorder in patients with 22q11.2 deletion syndrome
Biallelic loss‐of‐function variants of <i>EZH1</i> cause a novel developmental disorder with central precocious puberty
Hemiarhinia caused by a missense variation in <scp><i>SMCHD1</i></scp>: A mild phenotype in the clinical spectrum of Bosma arhinia microphthalmia syndrome
A milder form of NSRP1‐associated neurodevelopmental disorder, caused by a missense variant in the nuclear localization signal
Expansion of the prenatal phenotype of <scp>Baraitser–Winter</scp> syndrome: Presentation of two cases of multiple congenital anomaly syndrome
Osteopathia striata with cranial sclerosis as a cancer predisposition syndrome: The first report of neuroblastoma and review of all cancers in <scp>OSCS</scp>
<scp><i>LZTFL1</i></scp>, a rare cause of Bardet–Biedl syndrome: A new patient with severe short stature and moderate intellectual disability, more than casual associations?
<scp><i>CASK</i></scp> pathogenic variant which expands the clinical spectrum for <scp>MICPCH</scp> syndrome in an adult patient
Exploring the diverse clinical and variant spectrum of <scp>CEP78</scp>‐associated syndrome: Novel pathogenic variants identified in a case series
Emergence of the natural history of Myhre syndrome: 47 patients evaluated in the Massachusetts General Hospital Myhre Syndrome Clinic (2016–2023)
Rett syndrome diagnostic odyssey: Limitations of <scp>NextGen</scp> sequencing
Classification of isolated versus multiple birth defects: <scp>An</scp> automated process for population‐based registries
A rare case of uncharacterized autoinflammatory disease: Patient carrying variations in <i>NLRP3</i> and <i>TNFRSF1A</i> genes
Long‐term clinical observation of patients with heterozygous <scp><i>KIF1A</i></scp> variants
<scp>Dandy–Walker</scp> malformation in an individual with <i>ABL1</i> variant
Large regions of homozygosity in prenatal diagnosis
Large‐ and medium‐sized arterial aneurysms in two patients with <scp><i>SMAD4</i></scp>‐related juvenile polyposis syndrome
Neuropsychological functioning of adults with <scp><i>PTEN</i></scp> hamartoma tumor syndrome
Longitudinal adaptive behavioral outcomes in Ogden syndrome by seizure status and therapeutic intervention
Clinical and molecular characteristics of hemophilia <scp>A</scp> affected individuals and carriers: A 24 years experience from three centers
Adults with paternal <scp>UPD14</scp> causing <scp>Kagami–Ogata</scp> syndrome: Case report and review of the literature
Noonan syndrome‐like phenotype associated with an <i>ERF</i> frameshift variant
Trends in the survival of patients with trisomy 13 from 1995 to 2021: A population study in Japan
Filippi syndrome: Three new families suggest that urinary system abnormalities may belong to clinical spectrum of the disease
Aminotransferase trends in propionic acidemia
Next‐generation phenotyping in Nigerian children with Cornelia de Lange syndrome
Expanded prenatal phenotype of ALG12‐associated congenital disorder of glycosylation including bilateral multicystic kidneys
Clinical utility of comprehensive gene panel testing for common and rare causes of skeletal dysplasia and other skeletal disorders: Results from the largest cohort to date
Patient with a heterozygous pathogenic variant in <scp><i>CSNK2A1</i></scp> gene: A new case to update the <scp>Okur–Chung</scp> neurodevelopmental syndrome
<i>ITPR1</i>: The missing gene in miosis–ataxia syndrome?
Neurodevelopmental disorder associated with gene <i>ARF3</i>: A case report
Massive pericardial effusion in an infant with <scp>Aymé–Gripp</scp> syndrome: A case report and review of the literature
Exploring metabolic alterations in <scp>PYCR2</scp> deficiency: Unveiling pathways and clinical presentations of hypomyelinating leukodystrophy 10
Primary amenorrhea in myotonic dystrophy type 1: Initial presentation versus incidental finding on whole genome sequencing
<scp><i>SMAD4</i></scp> mosaicism in juvenile polyposis: Essential contribution of somatic analysis in diagnosis
X‐linked genetic associations in sporadic thoracic aortic dissection European descent
Decoding and Modeling the Human Immune System
In This Issue
Katherine M. Hyland, PhD
Table of Contents, Volume 194A, Number 6, June 2024
Expansion of the core features of <scp>VACTERL</scp> association to include genital anomalies
Cover Image, Volume 194A, Number 6, June 2024
The interplay of psychosis and non‐compliance with fatal outcome in an adult with <scp>MSUD</scp>
A novel <scp><i>AP1S2</i></scp> variant causing leaky splicing in X‐linked intellectual disability: Further delineation and intrafamilial variability Thai family
Different fetal effects on fingers from exposure to phenytoin, phenobarbital, and carbamazepine
A case report of a patient with neurodevelopmental disorder with impaired speech and hyperkinetic movements: A biallelic variant in the <i>ZNF142</i> gene
Mitochondrial phosphate‐carrier deficiency mimicking infantile‐onset Pompe disease
Qualitative and quantitative analysis of <i>MED12</i> c.887G&gt;A causing both missense and splicing variants in X‐linked Ohdo syndrome
Identification of a de novo <i>PUF60</i> variant associated with craniofacial microsomia
Phenotypic consequences of <i>GBA1</i> pathological variant <scp>R463C</scp> (p.<scp>R502C</scp>)
A novel <scp><i>GRK2</i></scp> variant in a patient with Jeune asphyxiating thoracic dysplasia accompanied by Morgagni hernia
Clinical case report of intractable paroxysmal sympathetic hyperactivity in <scp>TANGO2</scp> deficiency disorder
Further characterization of <scp>ARSK</scp>‐related mucopolysaccharidosis type 10
Immunological and hematological findings as major features in a patient with a new germline pathogenic <scp><i>CBL</i></scp> variant
Parents of children with Down syndrome reflect on their postnatal diagnoses, 2003–2022
Similarity of aortic events between siblings with heritable thoracic aortic diseases: Clinical analysis focusing on identical twins and <scp>same‐sex</scp> siblings
Laryngeal clefts in Prader–Willi syndrome: Feeding difficulties and aspiration not always caused by hypotonia
Novel <i>Alu</i> insertion in the <scp><i>ZEB2</i></scp> gene causing <scp>Mowat‐Wilson</scp> syndrome
“Why did I choose genetics?”: A survey of current and recent medical genetics and genomics residents provides insight into recruitment efforts
Ocular manifestations of <scp>CHARGE</scp> syndrome in a pediatric cohort with genotype/phenotype analysis
Alphamissense Predictive of Pathogenic Protein Variants
In Memoriam: Ishwar Chander Verma, MD (1936‐2024)
Table of Contents, Volume 194A, Number 5, May 2024
In This Issue
Cover Image, Volume 194A, Number 5, May 2024
Identification of a novel phenotype of external ear deformity related to <scp>Coffin–Siris</scp> syndrome‐9 and literature review
A novel 3q interstitial deletion including <i>GATA2</i> and <i>ZNF148</i>: A case report
Neurodevelopmental disorder in a patient with <scp><i>HMBS</i></scp> and <scp><i>SCN3A</i></scp> variants—A possibly blended phenotype further delineating autosomal recessive <scp><i>HMBS</i></scp> re
<scp>ECEL1</scp> related distal arthrogryposis <scp>5D</scp> in an Indian cohort—Report of recognizable musculoskeletal phenotype and a possible founder variant
Homozygosity for <scp>disease‐causing</scp> variants in <scp><i>AMT</i></scp> and <scp><i>GLDC</i></scp> in a patient with severe nonketotic hyperglycinemia
Truncating variants of the sterol recognition region of <i>SHH</i> cause hypertelorism phenotype rather than hypotelorism‐holoprosencephaly
Two sisters with RSPRY1‐related spondyloepimetaphyseal dysplasia
<scp><i>SYNGAP1</i></scp>‐related developmental and epileptic encephalopathy: Genotypic and phenotypic characteristics and longitudinal insights Korean
The society for craniofacial genetics and developmental biology 46th annual meeting
Clinical outcomes and medical management of achondroplasia in <scp>Japanese</scp> children: A retrospective medical record review of clinical data
Prenatal findings in 11 cases with craniofacial microsomia using the Alberta Congenital Anomalies Surveillance System, 1997–2019
Novel biallelic <scp><i>ZNF335</i></scp> variant causing primary microcephaly: <scp>A</scp> case report and radiological review Nepalese ancestry
Importance of the biochemical investigations for the functional characterization of a <scp><i>NPC1</i></scp> variant identified by exome sequencing
A case report of multicentric carpotarsal osteolysis syndrome: Depiction of a debilitating disease course
Familial recurrence of incontinentia pigmenti due to de novo pathogenic variants in the <i>IKBKG</i> gene
When rare meets common: Treatable genetic diseases are enriched in the general psychiatric population
Diagnostic findings and yield of investigations for children with developmental regression
A de novo frameshift variant in <scp><i>MED13</i></scp> gene in a patient with autism spectrum disorder and magnetic resonance imaging abnormalities mimicking tuberous sclerosis
Anxiety in Turner syndrome: Engaging community to address barriers and facilitators to diagnosis and care
Developmental and behavioral phenotypes of pediatric patients with <scp><i>PTEN</i></scp> hamartoma tumor syndrome
Patient experiences of receiving a diagnosis of hypermobile <scp>Ehlers–Danlos</scp> syndrome
<scp>RNA</scp> analysis and computer‐aided facial phenotyping help to classify a novel <i>TRIO</i> splice site variant
<i>ELMO2</i> biallelic pathogenic variants in a patient with gingival hypertrophy and cherubism phenotype: Case report and molecular review
Medical findings and congenital anomalies in <scp>Vermeer's</scp> paintings
Resolution of severe neurobehavioral difficulties in an individual with Primrose syndrome with sertraline
Early‐onset <scp>West</scp> syndrome with developmental delay associated with a novel <i>KLHL20</i> variant
Undiagnosed Disease Network collaborative approach in diagnosing rare disease in a patient with a mosaic <scp><i>CACNA1D</i></scp> variant
Clinical and molecular cytogenetic studies of five new patients with 20q11q12 deletion and review of the literature: Proposition of two critical regions
A further case of chondrodysplasia with growth failure occurring after hematopoietic stem cell transplantation (<scp>HSCT</scp>)
14q22.3 duplication including <scp><i>OTX2</i></scp> in a girl with medulloblastoma: A case report with literature review
Novel biallelic <i>SASS6</i> variants associated with primary microcephaly and fetal growth restriction
Next generation sequencing reveals novel compound heterozygous deletions in <i>NDUFAF2</i> in a child with mitochondrial complex I deficiency, nuclear type 10
Estimation of carrier frequencies utilizing the <scp>gnomAD</scp> database for <scp>ACMG</scp> recommended carrier screening and <scp>Finnish</scp> disease heritage conditions in non‐<scp>Finnish Euro non‐Finnish European; Finnish; Ashkenazi Jewish
First Gene Therapies Approved for Sickle Cell Disease
In This Issue
Table of Contents, Volume 194A, Number 4, April 2024
Megalencephaly secondary to a novel germline missense variant <scp>p.Asp322Tyr</scp> in <i>AKT3</i> associated with growth hormone deficiency and central hypothyroidism: A case report
Three Genes Associated with Neurodevelopmental Disorders Identified
Intracranial calcifications simulating <scp>Aicardi‐Goutières</scp> syndrome in <scp><i>PARS2</i></scp>‐related mitochondrial disease
<scp>LUMBAR syndrome–OEIS</scp> complex overlap: A case series and review
Taking the risk. A systematic review of ethical reasons and moral arguments in the clinical use of polygenic risk scores
<scp><i>TECPR2</i></scp>‐related hereditary sensory and autonomic neuropathy in two siblings from Palestine individuals of European ancestry; Palestinian siblings; Palestinian populations
Unraveling a history of overlap: A phenotypic comparison of <scp><i>RBCK1</i></scp>‐related disease and glycogen storage disease type <scp>IV</scp>
Communication of individuals with <scp>CDKL5</scp> deficiency disorder as observed by caregivers: A descriptive qualitative study
Rare predicted deleterious <scp><i>FEZF2</i></scp> variants are associated with a neurodevelopmental phenotype
<i>SMC1A</i> epilepsy syndrome: clinical data from a large international cohort
3q29 duplications: A cohort of 46 patients and a literature review
De novo variants predicting haploinsufficiency for <scp><i>DIP2C</i></scp> are associated with expressive speech delay
Hematologic malignancies in <scp>Li–Fraumeni</scp> syndrome: A case report
<i>RET</i> 634 germline/gonadal mosaicism generating a second pathogenic amino acid change in multiple endocrine neoplasia type 2A
Identification of a novel splice‐site <i>WWOX</i> variant with paternal uniparental isodisomy in a patient with infantile epileptic encephalopathy
Biallelic <scp><i>OTUD6B</i></scp> variants associated with a Kabuki syndrome‐like disorder in three siblings: A clinical report and literature review
Growth charts for weight and height of Indian children with Down syndrome
Metastatic adrenal gland neuroblastoma in an infant with trisomy 18: A case report
Common epilepsy variants from the general population are not associated with epilepsy among individuals with tuberous sclerosis complex
Experience with cascade screening: A comprehensive family pedigree analysis of two index patients with Fabry disease
<scp>PRKACA</scp>‐related, atrial defects‐polydactyly‐multiple congenital malformation syndrome in an Indian patient Indian
A case report on deficiency of adenosine deaminase 2 with relapse–remission course and analysis of genotype–phenotype correlation
Identification of novel <i>MYH14</i> variants in families with autosomal dominant sensorineural hearing loss
Australian researcher's perspectives on the Australian industry‐led moratorium on genetic tests in life insurance
Monodactyly in a patient with <scp>CHARGE</scp> syndrome: An additional case report
<scp><i>EFEMP1</i></scp> haploinsufficiency causes a Marfan‐like hereditary connective tissue disorder
<scp><i>RMRP</i></scp>‐related short stature: A report of six additional Japanese individuals with cartilage hair hypoplasia and literature review Finnish, Amish, Japanese, Japanese individuals, Finnish individuals, Amish populations
Susan D. Klugman, MD, FACOG, FACMG
Table of Contents, Volume 194A, Number 3, March 2024
In This Issue
Personal journeys to and in human genetics and dysmorphology
Cerebellar heterotopia in an 11‐year‐old child with <scp><i>KDM6B</i></scp>‐related neurodevelopmental disorder: A case report and review of the literature
Erratum to “De novo missense variants in <scp><i>ZBTB47</i></scp> are associated with developmental delays, hypotonia, seizures, gait abnormalities, and variable movement abnormalities”
First‐trimester noninvasive prenatal diagnosis of seven facioscapulohumeral muscular dystrophy type 1 families using <scp>SNP</scp>‐based amplicon sequencing: An earlier, rapid and safer way
Prevalence and descriptive epidemiology of choanal atresia and stenosis in Texas, 1999–2018
Development of specific growth charts for children with Fanconi anemia
The impact of a virtual mind–body program on symptoms of depression and anxiety among international <scp>English</scp>‐speaking adults with neurofibromatosis
Epilepsy with faint capillary malformation or reticulated telangiectasia associated with mosaic <i>AKT3</i> pathogenic variants
Expanding the phenotype of <scp><i>UPF3B</i></scp>‐related disorder: <scp>Case</scp> reports and literature review
A longitudinal characterization of the adaptive and behavioral profile in Sotos syndrome
<i>PUF60</i> loss‐of‐function with normal cognition should be considered in the differential diagnosis of Klippel–Feil syndrome
Feeding tube use and complications in <scp>Prader‐Willi</scp> syndrome: Data from the <scp>Global Prader‐Willi Syndrome Registry</scp>
Unraveling the molecular diagnosis of metaphyseal enchondromatosis with <scp>D‐2‐hydroxyglutaric</scp> aciduria: A <scp>22‐year</scp> quest
Medulloblastoma and other neoplasms in patients with heterozygous germline <scp><i>SUFU</i></scp> variants: A scoping review
Epilepsy in Legius syndrome: Coincidence or causation?
Early diagnostic clues of mucolipidosis type II: Significance of radiological findings
Naturally occurring splice variants dissect the functional domains of <scp>BHC80</scp> and emphasize the need for <scp>RNA</scp> analysis
Whole genome sequencing as a first‐tier diagnostic test for infants in neonatal intensive care units: <scp>A</scp> pilot study in <scp>Brazil</scp>
<scp>ARID2</scp>, a milder cause of <scp>Coffin‐Siris Syndrome</scp>? Broadening the phenotype with 17 additional individuals
Corrigendum to “Gain‐of‐function pathogenic variants in <scp><i>SMAD4</i></scp> are associated with neoplasia in Myhre syndrome”
X‐linked congenital adrenal hypoplasia: Report of long clinical follow‐up and description of a new complex variant in the <i>NR0B1</i> gene
Data from electronic healthcare records expand our understanding of X‐linked genetic diseases
Metabolic and other morbid complications in congenital generalized lipodystrophy type 4
Complex balanced intrachromosomal rearrangement involving <scp><i>PITX2</i></scp> identified as a cause of <scp>Axenfeld‐Rieger</scp> Syndrome
Understanding chronic pain in <scp>Neurofibromatosis Type 1</scp> using the <scp>Neurofibromatosis Pain Module</scp> (<scp>NFPM</scp>)
Progressive encephalopathy after routine 4‐month immunizations in a patient with <i>NAXD</i> genetic variant
Evaluation of functionality‐mobility in patients with skeletal dysplasias. Application of the STEMS tool (“everyday symptoms and mobility screening tool for skeletal dysplasias”)
Table of Contents, Volume 194A, Number 2, February 2024
David W. Smith Workshop: 44 Years and Going Strong
The Legacy of David W. Smith
In This Issue
Extended phenotypic characterization of a novel <scp>Helsmoortel‐van der Aa</scp> syndrome case series
Recurrent missense variant identified in two unrelated families with <scp><i>MPZL2</i></scp>‐related hearing loss, expanding the variant spectrum associated with <scp>DFNB111</scp>
Novel <scp><i>CHRNA3</i></scp> variants identified in a patient with bladder dysfunction, dysautonomia, and gastrointestinal dysmotility
An unusual diagnosis of alpha‐mannosidosis with ocular anomalies: Behind the scenes of a hidden copy number variation
Expanding the phenotypic spectrum for <i>CDK8</i>‐related disease: A case report
The first Turkish family with a novel biallelic missense variant of the <i>ALKBH8</i> gene: A study on the clinical and variant spectrum of <i>ALKBH8</i>‐related intellectual developmental disorders Turkish
c.202_204del in <scp><i>NUP214</i></scp> causes late onset form of febrile encephalopathy
Fragile X syndrome in the largest world clustering. I. Genetic epidemiology and founder effect outline
Novel molecular mechanism in Malan syndrome uncovered through genome sequencing reanalysis, exon‐level Array, and <scp>RNA</scp> sequencing
Further evidence that the neurodevelopmental gene <scp><i>FBXW7</i></scp> predisposes to <scp>Wilms</scp> tumor
Split‐hand/foot malformation 3 resulting from microduplications in 10q24 region in five patients from India Indian
Safety and tolerability of intravenous immunoglobulin infusion in Down syndrome regression disorder
Applying data science methodologies with artificial intelligence variant reinterpretation to map and estimate genetic disorder prevalence utilizing clinical data
Diagnosis of <i>TET3</i>‐Related Beck–Fahrner Syndrome in an Individual With Chorioretinal and Iris Colobomata Using a <scp>DNA</scp> Methylation Signature
Specialty Care Oases: Utilizing Geographic Information Systems to Evaluate Specialty Care Access for Individuals With Down Syndrome
Novel Digital Anomalies, Hippocampal Atrophy, and Mutations Expand the Genotypic and Phenotypic Spectra of <scp><i>CNKSR2</i></scp> in the Houge Type of X‐Linked Syndromic Intellectual Development Dis
Familial <i>RPL26</i> Variant Causing Congenital Anomalies Without Hematological Features of Diamond Blackfan Anemia
A Deep Intronic Splice Variant in <i>COL1A1</i> Causing Osteogenesis Imperfecta Type <scp>II</scp>
Nonrecurrent Triplication of 5q21.3q23.3: A Case Report and Review of the Literature
Identification of a Founder <scp><i>GLDN</i></scp> Variant Associated With “Lethal” Arthrogryposis in Nunavik Inuit: Implications for Obstetrical and Long‐Term Survivors' Management
Expansion of the Phenotype of You‐Hoover‐Fong Syndrome and Possible Increased Risk of Cancer
Recurrent <scp><i>FLNA</i> p.Gly1554Arg</scp> Variant Associated With Familial Ebstein Anomaly and Joint Stiffness
Prenatal Diagnosis of Proteus Syndrome: About a Case
Sodium Oxybate‐Treated Familial Myoclonus‐Dystonia Syndrome Due to Novel <scp><i>SGCE</i></scp> Variant