Journal of Medical Genetics - 2008

143 articles | Last updated: 2025-12-03 14:12:57
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T A T A T A T A
Novel POLG1 mutations associated with neuromuscular and liver phenotypes in adults and children
Novel mutations in patients with McArdle disease by analysis of skeletal muscle mRNA
Succinate dehydrogenase subunit B (SDHB) gene deletion associated with a composite paraganglioma/neuroblastoma
Mutations in LAMB2 causing a severe form of synaptic congenital myasthenic syndrome
Correction
Investigation of somatic NKX2-5 mutations in congenital heart disease
Genomic rearrangements in OPA1 are frequent in patients with autosomal dominant optic atrophy
Correction
NARP syndrome in a patient harbouring an insertion in the MT-ATP6 gene that results in a truncated protein
Familial occurrence of schwannomas and malignant rhabdoid tumour associated with a duplication in SMARCB1
Recessive osteogenesis imperfecta caused by LEPRE1 mutations: clinical documentation and identification of the splice form responsible for prolyl 3-hydroxylation
Epigenetic mutations of the imprinted IGF2-H19 domain in Silver-Russell syndrome (SRS): results from a large cohort of patients with SRS and SRS-like phenotypes
Bone health and fracture rate in individuals with neurofibromatosis 1 (NF1)
Corrections
No reason yet to change diagnostic criteria for Noonan, Costello and cardio-facio-cutaneous syndromes
Corrections
Genetics of infantile seizures with paroxysmal dyskinesia: the infantile convulsions and choreoathetosis (ICCA) and ICCA-related syndromes
Genomic duplication resulting in increased copy number of genes encoding the sister chromatid cohesion complex conveys clinical consequences distinct from Cornelia de Lange
The molecular landscape of ASPM mutations in primary microcephaly
Polymorphisms in C2, CFB and C3 are associated with progression to advanced age related macular degeneration associated with visual loss
Detection of cryptic pathogenic copy number variations and constitutional loss of heterozygosity using high resolution SNP microarray analysis in 117 patients referred for cytogenetic analysis and imp
Inherited mitochondrial optic neuropathies
In vitro splicing analysis showed that availability of a cryptic splice site is not a determinant for alternative splicing patterns caused by +1G->A mutations in introns of the dystrophin gene
Risk reducing mastectomy: outcomes in 10 European centres
Clinical and genetic delineation of neurodegeneration with brain iron accumulation
Clinical and molecular progress in hereditary paraganglioma
Correction
Phenotypic variability among patients with hyperornithinaemia-hyperammonaemia-homocitrullinuria syndrome homozygous for the delF188 mutation in SLC25A15
Ring syndrome: still true?
Towards more effective and equitable genetic testing for BRCA1 and BRCA2 mutaion carriers
Intergenerational CAG repeat instability is highly heritable in Huntington's disease
Novel transglutaminase-1 mutations and genotype-phenotype investigations of 104 patients with autosomal recessive congenital ichthyosis in the USA
Intranasal insulin to improve developmental delay in children with 22q13 deletion syndrome: an exploratory clinical trial
Longitudinal study of neurofibromatosis 1 associated plexiform neurofibromas
Effect of CHEK2 missense variant I157T on the risk of breast cancer in carriers of other CHEK2 or BRCA1 mutations
Spectrum of SCN1A gene mutations associated with Dravet syndrome: analysis of 333 patients
Contribution of TARDBP mutations to sporadic amyotrophic lateral sclerosis
Chromosome 15q11-13 duplication syndrome brain reveals epigenetic alterations in gene expression not predicted from copy number
Minocycline promotes dendritic spine maturation and improves behavioural performance in the fragile X mouse model
A genome-wide association study identifies an association of a common variant in TERT with susceptibility to idiopathic pulmonary fibrosis
Early onset of hypokalaemic periodic paralysis caused by a novel mutation of the CACNA1S gene
Is genetics inhumane?
2q23.1 microdeletion identified by array comparative genomic hybridisation: an emerging phenotype with Angelman-like features?
20p12.3 microdeletion predisposes to Wolff-Parkinson-White syndrome with variable neurocognitive deficits
Clinical experience in the evaluation of 30 patients with a prior diagnosis of FG syndrome
Six new coeliac disease loci replicated in an Italian population confirm association with coeliac disease
Site-dependent differences in both prelamin A and adipogenic genes in subcutaneous adipose tissue of patients with type 2 familial partial lipodystrophy
Microdeletion/duplication at 15q13.2q13.3 among individuals with features of autism and other neuropsychiatric disorders
Schimke immuno-osseous dysplasia: SMARCAL1 loss-of-function and phenotypic correlation
Mutation in the HPGD gene encoding NAD+ dependent 15-hydroxyprostaglandin dehydrogenase underlies isolated congenital nail clubbing (ICNC)
Association of a null allele of SPRN with variant Creutzfeldt-Jakob disease
Functional polymorphisms in the BRCA1 promoter influence transcription and are associated with decreased risk for breast cancer in Chinese women
Phenotypic variation within European carriers of the Y-chromosomal gr/gr deletion is independent of Y-chromosomal background European carriers
Identification of a novel TP53 germline mutation E285V in a rare case of paediatric adrenocortical carcinoma and choroid plexus carcinoma
GJB2 and hearing impairment: promoter defects do not explain the excess of monoallelic mutations
Skewed X inactivation is associated with phenotype in a female with adrenal hypoplasia congenita
Correction
Dynamic CpG methylation of the KCNQ1OT1 gene during maturation of human oocytes
Further delineation of Pitt-Hopkins syndrome: phenotypic and genotypic description of 16 novel patients
Refinement of cortical dysgeneses spectrum associated with TUBA1A mutations
Autistic phenotypes and genetic testing: state-of-the-art for the clinical geneticist
Association of haplotypes spanning PDZ-GEF2, LOC728637 and ACSL6 with schizophrenia in Han Chinese
High frequency of submicroscopic genomic aberrations detected by tiling path array comparative genome hybridisation in patients with isolated congenital heart disease
A novel mutation in the sulfate transporter gene SLC26A2 (DTDST) specific to the Finnish population causes de la Chapelle dysplasia Finnish population
A PCSK9 variant and familial combined hyperlipidaemia
Reversible phenotype in a mouse model of Hutchinson-Gilford progeria syndrome
Polymorphisms in the C-type lectin genes cluster in chromosome 19 and predisposition to severe acute respiratory syndrome coronavirus (SARS-CoV) infection
The HBS1L-MYB intergenic region on chromosome 6q23 is a quantitative trait locus controlling fetal haemoglobin level in carriers of  -thalassaemia
Mutations in JARID1C are associated with X-linked mental retardation, short stature and hyperreflexia
DNA methylation in intron 1 of the frataxin gene is related to GAA repeat length and age of onset in Friedreich ataxia patients
The terminal 760 kb region on 4p16 is unlikely to be the critical interval for growth delay in Wolf-Hirschhorn syndrome
Congenital heart disease is a feature of severe infantile spinal muscular atrophy
Epigenetic analysis of the critical region I for premature ovarian failure: demonstration of a highly heterochromatic domain on the long arm of the mammalian X chromosome
Clinical and molecular delineation of the 17q21.31 microdeletion syndrome
Mapping of 5q35 chromosomal rearrangements within a genomically unstable region
Cerebro-oculo-facio-skeletal syndrome: three additional cases with CSB mutations, new diagnostic criteria and an approach to investigation
Alteration of expression of muscle specific isoforms of the fragile X related protein 1 (FXR1P) in facioscapulohumeral muscular dystrophy patients
The CTG repeat expansion size correlates with the splicing defects observed in muscles from myotonic dystrophy type 1 patients
Biallelic loss of function of the promyelocytic leukaemia zinc finger (PLZF) gene causes severe skeletal defects and genital hypoplasia
Pseudomitochondrial genome haunts disease studies
Neurodevelopmental abnormalities associated with severe congenital neutropenia due to the R86X mutation in the HAX1 gene
ADVIRC is caused by distinct mutations in BEST1 that alter pre-mRNA splicing
Heart-hand syndrome of Slovenian type: a new kind of laminopathy Slovenian
Trichothiodystrophy: a systematic review of 112 published cases characterises a wide spectrum of clinical manifestations
Comparison of predictive models, clinical criteria and molecular tumour screening for the identification of patients with Lynch syndrome in a population-based cohort of colorectal cancer patients
Genetic analysis of 56 polymorphisms in 17 genes involved in methionine metabolism in patients with abdominal aortic aneurysm
Identification of new mutations in the ETHE1 gene in a cohort of 14 patients presenting with ethylmalonic encephalopathy
Lack of C20orf133 and FLRT3 mutations in 43 patients with Kabuki syndrome in Japan
Recurrent reciprocal deletions and duplications of 16p13.11: the deletion is a risk factor for MR/MCA while the duplication may be a rare benign variant
Clinical and molecular aspects of RAS related disorders
New SMS mutation leads to a striking reduction in spermine synthase protein function and a severe form of Snyder-Robinson X-linked recessive mental retardation syndrome
Prevalence and heritability of the metabolic syndrome and its individual components in a Dutch isolate: the Erasmus Rucphen Family study Dutch isolate
Bitter taste receptor gene polymorphisms are an important factor in the development of nicotine dependence in African Americans African Americans
Complex and segmental uniparental disomy updated
Coexistence of mutations in PINK1 and mitochondrial DNA in early onset parkinsonism
Heme oxygenase 1 variations and lung function decline in smokers: proof of replication
Mosaicism in sporadic neurofibromatosis type 1: variations on a theme common to other hereditary cancer syndromes?
A novel A121T mutation in human cationic trypsinogen associated with hereditary pancreatitis: functional data indicating a loss-of-function mutation influencing the R122 trypsin cleavage site
Molecular basis of the Li-Fraumeni syndrome: an update from the French LFS families
Mosaic maternal uniparental disomy of chromosome 11 in a patient with Silver-Russell syndrome
Dosage compensation of the mammalian X chromosome influences the phenotypic variability of X-linked dominant male-lethal disorders
Dentin phosphoprotein frameshift mutations in hereditary dentin disorders and their variation patterns in normal human population
High frequency of submicroscopic chromosomal imbalances in patients with syndromic craniosynostosis detected by a combined approach of microsatellite segregation analysis, multiplex ligation-dependent
Termination of damaged protein repair defines the occurrence of symptoms in carriers of the m.3243A>G tRNALeu mutation
Noonan and cardio-facio-cutaneous syndromes: two clinically and genetically overlapping disorders
Contribution of PTEN large rearrangements in Cowden disease: a multiplex amplifiable probe hybridisation (MAPH) screening approach
Towards more effective and equitable genetic testing for BRCA1 and BRCA2 mutation carriers
Double outlet right ventricle: aetiologies and associations
We need a detailed phenome in the phenomenon of genetics and congenital heart disease
Identification of a 2244 base pair interstitial deletion within the human ESR1 gene in the Spanish population Spanish population
Genome-wide linkage scan for loci of musical aptitude in Finnish families: evidence for a major locus at 4q22
Screening BRCA1 and BRCA2 unclassified variants for splicing mutations using reverse transcription PCR on patient RNA and an ex vivo assay based on a splicing reporter minigene
Predicting the likelihood of carrying a BRCA1 or BRCA2 mutation: validation of BOADICEA, BRCAPRO, IBIS, Myriad and the Manchester scoring system using data from UK genetics clinics
X-linked ichthyosis (steroid sulfatase deficiency) is associated with increased risk of attention deficit hyperactivity disorder, autism and social communication deficits
The fragile X prevalence paradox
Expansion in size of a terminal deletion: a paradigm shift for parental follow-up studies
Detection of known and novel genomic rearrangements by array based comparative genomic hybridisation: deletion of ZNF533 and duplication of CHARGE syndrome genes
Meta-analysis of vascular endothelial growth factor variations in amyotrophic lateral sclerosis: increased susceptibility in male carriers of the -2578AA genotype
Screening for familial ovarian cancer: poor survival of BRCA1/2 related cancers
Triphalangeal thumb-polysyndactyly syndrome and syndactyly type IV are caused by genomic duplications involving the long range, limb-specific SHH enhancer
Disruption of contactin 4 in three subjects with autism spectrum disorder
Heterogeneity in the processing defect of SLC26A4 mutants
A systematic review of the clinical validity and clinical utility of DNA testing for hereditary haemochromatosis type 1 in at-risk populations
Contribution of molecular analyses in diagnosing Marfan syndrome and type I fibrillinopathies: an international study of 1009 probands
Identifying pathogenic genetic background of simplex or multiplex retinitis pigmentosa patients: a large scale mutation screening study
Molecular characterisation of SMARCB1 and NF2 in familial and sporadic schwannomatosis
Identification of a novel PEX14 mutation in Zellweger syndrome
A missense mutation in PTCH2 underlies dominantly inherited NBCCS in a Chinese family
High frequency of genomic deletions--and a duplication--in the LIS1 gene in lissencephaly: implications for molecular diagnosis
The protective effect of farm animal exposure on childhood allergy is modified by NPSR1 polymorphisms
Interferon regulatory factor 5 (IRF5) gene variants are associated with multiple sclerosis in three distinct populations
BHD mutations, clinical and molecular genetic investigations of Birt-Hogg-Dube syndrome: a new series of 50 families and a review of published reports
CYLD mutations in familial skin appendage tumours
Deletion of C2orf34, PREPL and SLC3A1 causes atypical hypotonia-cystinuria syndrome
Detection of early FXTAS motor symptoms using the CATSYS computerised neuromotor test battery
Recent advances in the molecular pathology, cell biology and genetics of ciliopathies
Comparison of targeted and whole genome analysis of postnatal specimens using a commercially available array based comparative genomic hybridisation (aCGH) microarray platform
Clinical and molecular characteristics of 1qter microdeletion syndrome: delineating a critical region for corpus callosum agenesis/hypogenesis
A frame-shift mutation of PMS2 is a widespread cause of Lynch syndrome
Genome-wide linkage scan for maximum and length-dependent knee muscle strength in young men: significant evidence for linkage at chromosome 14q24.3
CDKN2A mutations and melanoma risk in the Icelandic population
A microduplication of the long range SHH limb regulator (ZRS) is associated with triphalangeal thumb-polysyndactyly syndrome
Comparison of X-chromosome inactivation patterns in multiple tissues from human females