A guide for the diagnosis of rare and undiagnosed disease: beyond the exome

S Marwaha, JW Knowles, EA Ashley - Genome medicine, 2022 - Springer
Rare diseases affect 30 million people in the USA and more than 300–400 million
worldwide, often causing chronic illness, disability, and premature death. Traditional …

Machine learning for integrating data in biology and medicine: Principles, practice, and opportunities

M Zitnik, F Nguyen, B Wang, J Leskovec… - Information …, 2019 - Elsevier
New technologies have enabled the investigation of biology and human health at an
unprecedented scale and in multiple dimensions. These dimensions include a myriad of …

PhenoScore quantifies phenotypic variation for rare genetic diseases by combining facial analysis with other clinical features using a machine-learning framework

AJM Dingemans, M Hinne, KMG Truijen, L Goltstein… - Nature Genetics, 2023 - nature.com
Several molecular and phenotypic algorithms exist that establish genotype–phenotype
correlations, including facial recognition tools. However, no unified framework that …

Generalisable long COVID subtypes: findings from the NIH N3C and RECOVER programmes

JT Reese, H Blau, E Casiraghi, T Bergquist… - …, 2023 - thelancet.com
Background Stratification of patients with post-acute sequelae of SARS-CoV-2 infection
(PASC, or long COVID) would allow precision clinical management strategies. However …

MutationTaster2021

R Steinhaus, S Proft, M Schuelke… - Nucleic Acids …, 2021 - academic.oup.com
Here we present an update to MutationTaster, our DNA variant effect prediction tool. The
new version uses a different prediction model and attains higher accuracy than its …

Expansion of the Human Phenotype Ontology (HPO) knowledge base and resources

S Köhler, L Carmody, N Vasilevsky… - Nucleic acids …, 2019 - academic.oup.com
Abstract The Human Phenotype Ontology (HPO)—a standardized vocabulary of phenotypic
abnormalities associated with 7000+ diseases—is used by thousands of researchers …

Semantic similarity and machine learning with ontologies

M Kulmanov, FZ Smaili, X Gao… - Briefings in …, 2021 - academic.oup.com
Ontologies have long been employed in the life sciences to formally represent and reason
over domain knowledge and they are employed in almost every major biological database …

The human phenotype ontology in 2017

S Köhler, NA Vasilevsky, M Engelstad… - Nucleic acids …, 2017 - academic.oup.com
Deep phenoty** has been defined as the precise and comprehensive analysis of
phenotypic abnormalities in which the individual components of the phenotype are observed …

Bioinformatics and computational tools for next-generation sequencing analysis in clinical genetics

R Pereira, J Oliveira, M Sousa - Journal of clinical medicine, 2020 - mdpi.com
Clinical genetics has an important role in the healthcare system to provide a definitive
diagnosis for many rare syndromes. It also can have an influence over genetics prevention …

Comparison and integration of deleteriousness prediction methods for nonsynonymous SNVs in whole exome sequencing studies

C Dong, P Wei, X Jian, R Gibbs… - Human molecular …, 2015 - academic.oup.com
Accurate deleteriousness prediction for nonsynonymous variants is crucial for distinguishing
pathogenic mutations from background polymorphisms in whole exome sequencing (WES) …