Mendelian inheritance revisited: dominance and recessiveness in medical genetics

J Zschocke, PH Byers, AOM Wilkie - Nature Reviews Genetics, 2023 - nature.com
Understanding the consequences of genotype for phenotype (which ranges from molecule-
level effects to whole-organism traits) is at the core of genetic diagnostics in medicine. Many …

Loss-of-function, gain-of-function and dominant-negative mutations have profoundly different effects on protein structure

L Gerasimavicius, BJ Livesey, JA Marsh - Nature communications, 2022 - nature.com
Most known pathogenic mutations occur in protein-coding regions of DNA and change the
way proteins are made. Taking protein structure into account has therefore provided great …

Insights into the ANKRD11 variants and short-stature phenotype through literature review and ClinVar database search

D He, M Zhang, Y Li, F Liu, B Ban - Orphanet Journal of Rare Diseases, 2024 - Springer
Ankyrin repeat domain containing-protein 11 (ANKRD11), a transcriptional factor
predominantly localized in the cell nucleus, plays a crucial role in the expression regulation …

Integrative proteomic characterization of adenocarcinoma of esophagogastric junction

S Li, L Yuan, ZY Xu, JL Xu, GP Chen, X Guan… - Nature …, 2023 - nature.com
The incidence of adenocarcinoma of the esophagogastric junction (AEG) has been rapidly
increasing in recent decades, but its molecular alterations and subtypes are still obscure …

Intrinsic endothelial hyperresponsiveness to inflammatory mediators drives acute episodes in models of Clarkson disease

AJ Ablooglu, WS Chen, Z ** using Interferometric Synthetic Aperture Radar (InSAR) and machine learning models in a semiarid region of Iran
H Gharechaee, AN Samani, SK Sigaroodi… - Land, 2023 - mdpi.com
Most published studies identify groundwater extraction as the leading cause of land
subsidence (LS). However, the causes of LS are not only attributable to groundwater …

Characterizing and explaining the impact of disease-associated mutations in proteins without known structures or structural homologs

N Sen, I Anishchenko, N Bordin, I Sillitoe… - Briefings in …, 2022 - academic.oup.com
Mutations in human proteins lead to diseases. The structure of these proteins can help
understand the mechanism of such diseases and develop therapeutics against them. With …

Understanding structure-guided variant effect predictions using 3D convolutional neural networks

G Ramakrishnan, C Baakman, S Heijl… - Frontiers in molecular …, 2023 - frontiersin.org
Predicting pathogenicity of missense variants in molecular diagnostics remains a challenge
despite the available wealth of data, such as evolutionary information, and the wealth of …

Proteome-scale prediction of molecular mechanisms underlying dominant genetic diseases

M Badonyi, JA Marsh - Plos one, 2024 - journals.plos.org
Many dominant genetic disorders result from protein-altering mutations, acting primarily
through dominant-negative (DN), gain-of-function (GOF), and loss-of-function (LOF) …

VPatho: a deep learning-based two-stage approach for accurate prediction of gain-of-function and loss-of-function variants

F Ge, C Li, S Iqbal, A Muhammad, F Li… - Briefings in …, 2023 - academic.oup.com
Determining the pathogenicity and functional impact (ie gain-of-function; GOF or loss-of-
function; LOF) of a variant is vital for unraveling the genetic level mechanisms of human …