Genodermatoses, a group of inherited skin disorders, are characterized by significant genetic heterogeneity and clinical variability, often posing diagnostic and therapeutic challenges. Advances in next-generation sequencing (NGS) technologies, such as whole exome sequencing (WES) and clinical exome sequencing (CES), have transformed the diagnostic landscape by enabling comprehensive genetic analysis. This study aimed to investigate the molecular spectrum and clinical relevance of genetic findings in 43 patients diagnosed with genodermatoses. Demographic, clinical, and molecular data were collected, and genetic testing was performed using the MGI-Seq platform. Variants were analyzed for pathogenicity, zygosity, and novelty. Neurofibromatosis Type 1 (27.9%) and Epidermolysis Bullosa (23.2%) were the most common diagnoses, followed by Ichthyosis (16.2%) and Oculocutaneous Albinism (13.9%). Less frequent conditions included Ectodermal Dysplasia (6.9%) and single cases of Palmoplantar Keratoderma, PTEN Hamartoma Syndrome, Rothmund-Thomson Syndrome, Xeroderma Pigmentosum, and Megaconial Congenital Muscular Dystrophy (each 2.3%). Molecular findings underscored the genetic complexity of genodermatoses, with 42 distinct variants identified across 19 genes. Of these, 13 variants (31%) were novel, expanding the known molecular spectrum. The novel variants were detected in genes including NF1, COL7A1, ITGB4, COL17A1, NIPAL4, ALOX12B, KRT10, ST14, OCA2, and PTEN, highlighting the diagnostic value of comprehensive genetic analysis. The mean age at diagnosis varied significantly among conditions, reflecting the diagnostic challenges and clinical variability of genodermatoses. This study emphasizes the critical role of WES and CES in diagnosing genodermatoses and understanding their molecular basis, which enhances diagnostic accuracy and supports personalized management strategies.
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http://dx.doi.org/10.1007/s00403-025-04056-7 | DOI Listing |
Eur J Neurosci
March 2025
School of Psychology, University of Auckland, Auckland, New Zealand.
The aim of the study was to determine the test-retest reliability of MMN and LDN recorded to simple speech contrasts in children with listening difficulties. MMN and LDN responses were recorded from Fz and Cz electrodes for a /da/-/ga/ contrast twice within a 10-day period. To extract MMN and LDN, auditory-evoked responses to /ga/ stimuli presented alone were subtracted from the responses to /ga/ presented within an oddball sequence.
View Article and Find Full Text PDFEBioMedicine
February 2025
Institute of Medical Sciences, School of Medicine, Medical Sciences & Nutrition, University of Aberdeen, Aberdeen, UK; Institute for Clinical Research and Systems Medicine, Health and Medical University, Potsdam, Germany.
Background: Maternal smoking and foetal exposure to nicotine and other harmful chemicals in utero remains a serious public health issue with little knowledge about the underlying genetics and consequences of maternal smoking in ageing individuals. Here, we investigated the epidemiology and genomic architecture of maternal smoking in a middle-aged population and compare the results to effects observed in the developing foetus.
Methods: In the current project, we included 351,562 participants from the UK Biobank (UKB) and estimated exposure to maternal smoking status during pregnancy through self-reporting from the UKB participants about the mother's smoking status around their birth.
Rev Environ Health
March 2025
Public Health Department- Dubai Health Authority, Dubai, United Arab Emirates.
Introduction: The developing brain, especially vulnerable during neuroplastic phases, is influenced by environmental and genetic factors. Understanding the impacts of air pollution on children's and young adults' mental health is an emerging research field.
Content: This review systematically examines the adverse associations of ambient air pollutants on mental health.
ESC Heart Fail
March 2025
Institute of Health Informatics Research, University College London, London, UK.
Aims: Atrial fibrillation (AF) is a frequent comorbidity in heart failure (HF). We analysed factors associated with new-onset atrial fibrillation in patients with heart failure using linked real-world UK data from primary and secondary care, along with findings from genome-wide association studies.
Methods And Results: Among 163 174 participants with a diagnosis of HF (January 1998 to May 2016) from Clinical Practice Research Datalink (CPRD) and Hospital Episodes Statistics (HES), 111 595 participants had no previous history of AF (mean age 76.
Ophthalmic Genet
March 2025
Ophthalmic Genetics & Visual Function Branch, National Eye Institute, Bethesda, Virginia, USA.
The development of the neural retina requires a complex, spatiotemporally regulated network of gene expression. Here we review the role of the cone rod homeobox () transcription factor in specification and differentiation of retinal photoreceptors and its function in inherited retinal diseases such as cone-rod dystrophy (CoRD), dominant retinitis pigmentosa (RP), and Leber's congenital amaurosis (LCA). We delineate the findings of animal models and, more recently, human retinal organoids in elucidating molecular mechanisms of CRX activity and the pathogenesis of inherited photoreceptor degenerations.
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