A call to action to scale up research and clinical genomic data sharing

Z Stark, D Glazer, O Hofmann, A Rendon… - Nature Reviews …, 2025 - nature.com
Genomic data from millions of individuals have been generated worldwide to drive discovery
and clinical impact in precision medicine. Lowering the barriers to using these data …

Advancing long-read nanopore genome assembly and accurate variant calling for rare disease detection

S Negi, SL Stenton, SI Berger, P Canigiula… - The American Journal of …, 2025 - cell.com
More than 50% of families with suspected rare monogenic diseases remain unsolved after
whole-genome analysis by short-read sequencing (SRS). Long-read sequencing (LRS) …

RNA splicing: a split consensus reveals two major 5′ splice site classes

MT Parker, SM Fica, GG Simpson - Open Biology, 2025 - royalsocietypublishing.org
The established consensus sequence for human 5′ splice sites masks the presence of two
major splice site classes defined by preferential base-pairing potentials with either U5 …

Genomic reanalysis of a pan-European rare-disease resource yields new diagnoses

S Laurie, W Steyaert, E de Boer, K Polavarapu… - Nature Medicine, 2025 - nature.com
Genetic diagnosis of rare diseases requires accurate identification and interpretation of
genomic variants. Clinical and molecular scientists from 37 expert centers across Europe …

Mutations in the U2 snRNA gene RNU2-2P cause a severe neurodevelopmental disorder with prominent epilepsy

D Greene, K De Wispelaere, J Lees, A Katrinecz… - medRxiv, 2024 - medrxiv.org
The major spliceosome comprises the five snRNAs U1, U2, U4, U5 and U6. We recently
showed that mutations in RNU4-2, which encodes U4 snRNA, cause one of the most …

De novo missense variants in the PP2A regulatory subunit PPP2R2B in a neurodevelopmental syndrome: potential links to mitochondrial dynamics and …

P Sandal, CJ Jong, RA Merrill… - Human molecular …, 2025 - academic.oup.com
The heterotrimeric protein phosphatase 2A (PP2A) complex catalyzes about half of Ser/Thr
dephosphorylations in eukaryotic cells. A CAG repeat expansion in the neuron-specific …

Rare developmental disorder caused by variants in a small RNA gene

H Heyne - 2024 - nature.com
Rare developmental disorder caused by variants in a small RNA gene Skip to main content
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Deep phenoty** of 11 individuals with pathogenic variants in RNU4-2 reveals a clinically recognizable syndrome

I Valenzuela, M Codina-Solà, E Vazquez… - Genetics in …, 2024 - Elsevier
Purpose Despite ever-increasing knowledge of the genetic etiologies of
neurodevelopmental disorders, approximately half remain undiagnosed after exome or …

ReNU syndrome–a newly discovered prevalent neurodevelopmental disorder

VF Burns, EJ Radford - Trends in Genetics, 2024 - cell.com
Two recent papers have identified genetic variants in the noncoding gene RNU4-2 to cause
a frequent neurodevelopmental disorder. This work will have a substantial impact on the …

GREGoR: Accelerating Genomics for Rare Diseases

M Dawood, B Heavner, MM Wheeler, RA Ungar… - arxiv preprint arxiv …, 2024 - arxiv.org
Rare diseases are collectively common, affecting approximately one in twenty individuals
worldwide. In recent years, rapid progress has been made in rare disease diagnostics due …