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Franke, L.

Publications and source records attributed to Franke, L..

At least 19 recordsLinked to original sources

High cell-type specificity of eQTLs revealed by single-nucleus analyses of brain and blood

Identifying causal mechanisms from genome-wide association studies (GWAS) requires an understanding of how disease-associated genetic variants influence gene expression in specific cell types. Here, we present scMetaBrain, a large-scale single-nucleus RNA-sequencing (snRNA-seq) resource derived using 1,260 samples from 785 individuals spanning 10 brain datasets. By analyzing 3.9 million transcriptomes, we identified 19,371 unique expression quantitative trait locus (eQTL) genes (eGenes) at a major cell type level, with the largest number of eQTLs observed in excitatory neurons. Notably, 31% of the eQTLs detected were highly cell-type-specific, with most restricted to excitatory neurons (69%). We compared the eQTLs with bulk RNA-seq datasets across different tissues and with a newly generated single nucleus dataset of 123 donors from peripheral blood mononuclear cells. We observed that differences in eQTL effect sizes between brain cell types are often as large as comparing eQTLs between brain tissue and non-brain tissue from bulk RNA-seq studies. Furthermore, we observe that eQTL effect size agreement was highest for cell types with similar function, even when comparing brain to blood cells. This suggests that that bulk analyses substantially overestimate eQTL agreement, likely due to tissue-level averaging of cellular regulatory effects. Through colocalization, we prioritized 662 genes for 11 brain-related traits and prioritized a single cell type in 68% of genes. Our findings demonstrate that eQTL effects are far more cell-type-specific than previously recognized, underscoring the need to expand single-cell eQTL studies across diverse tissues and cell types to fully capture the regulatory architecture of genetic variants.

genetics↗

Epigenome-wide association study meta-analysis of wellbeing

Wellbeing is associated with both behavioral phenotypes as well as several key life outcomes, such as health, employment, and coping with stressful events. These phenotypes associated with wellbeing could be potential indicators of differential epigenetic patterns between individuals that differ in their levels of wellbeing. We performed the largest epigenome-wide (EWAS) meta-analysis of wellbeing to date by combining whole blood DNA methylation data (Illumina 450k array) from 13 cohorts from Europe, Australia, and the USA (N = 10,757 participants). After correcting for smoking and BMI, no epigenome-wide significant methylation sites were identified. We tested whether a weighted methylation score (MS) based on leave-one-cohort-out EWAS meta-analysis summary statistics predicted wellbeing in an independent cohort, and whether prediction was significant over and above the polygenic score (PGS) for wellbeing. The MS was associated with wellbeing (variance explained=0.22%, p=0.03) and was no longer significant after adding the polygenic score (PGS; variance explained=0.43%, p=0.0046, MS; variance explained=0.07%, p=0.2842). We further compared DNA methylation levels in 16 pairs of monozygotic twins discordant for wellbeing. These analyses revealed no significant within-pair DNA methylation differences at the top-sites from the meta-analysis or in MS. Our results suggest that larger EWAS meta-analyses with uniform phenotype assessment are required to identify methylation sites associated with wellbeing.

genomics↗

Disease-associated variants are enriched for altering cell-type-specific gene co-expression relationships

Genes act within complex regulatory networks, and genetic variants can perturb these networks by altering gene co-expression. Here, we performed co-expression quantitative trait locus (co-eQTL) mapping using single-cell RNA-seq from the sc-eQTLGen consortium (1,330 donors, >2 million cells), enabling sensitive detection and prioritization of informative variant-gene-gene triplets. We identified co-eQTLs for 398 eGenes where a nearby genetic variant affected both the genes expression (cis-gene) and its co-expression with other genes, often implicating upstream regulators. For 181 genes, we inferred a likely upstream transcription factor, with motif disruption predicted for 41 genes. These upstream genes are more often loss-of-function intolerant and show more network connections, providing an explanation for why co-eQTL variants are 2.8x more strongly associated with immune diseases than classical eQTLs. These findings position co-eQTLs as mechanistic links between genetic variation and disease, revealing how variants can rewire cell-type-specific gene networks.

genetics↗

Widespread Distribution of α-Synuclein Oligomers in LRRK2-related Parkinson's Disease

Mutations in leucine-rich repeat kinase 2 (LRRK2) are the most common cause of familial and sporadic Parkinsons disease (PD). While the clinical features of LRRK2-PD patients resemble those of typical PD, there are significant differences in the pathological findings. The pathological hallmark of definite PD is the presence of -synuclein (SYN)-positive Lewy-related pathology; however, approximately half of LRRK2-PD cases do not have Lewy-related pathology. Lewy-related pathology is a late-stage SYN aggregation that can be visualized with hematoxylin and eosin stains or conventional immunohistochemistry (IHC). Increasing evidence has indicated that SYN oligomers, which represent the early-stage of SYN aggregation, may have neurotoxicity. Visualization of SYN oligomers requires specialized staining techniques, such as SYN-proximity ligation assay (PLA). The distribution and severity of SYN oligomers in the human brain of LRRK2-PD patients remain unknown. In this study, we performed phosphorylated SYN-IHC and SYN-PLA staining on postmortem brain sections of patients with three pathogenic LRRK2 mutants: p.G2019S (n=5), p.I2020T (n=5), and p.R1441C (n=4). The severity of Lewy-related pathology and SYN oligomers were assessed semi-quantitatively in the brainstem, limbic lobe, basal ganglia, and cerebral cortex. SYN oligomers were detected in LRRK2-PD cases even in cases without Lewy-related pathology; a negative correlation was observed between Lewy-related pathology and SYN oligomers (r=-0.26 [-0.39, -0.12]; P<0.0001). Our findings suggest that SYN oligomers may represent a common pathological feature of LRRK2-PD. Notably, patients harboring p.G2019S and p.I2020T had significantly higher levels of SYN oligomers in those without Lewy-related pathology compared to those with Lewy-related pathology. These cases also had a trend toward shorter disease duration. These results imply that in LRRK2-PD, SYN oligomers may initially accumulate in the brain but do not progress to form Lewy-related pathology. The present study suggests that targeting SYN oligomers may be a therapeutic strategy for LRRK2-PD even if there is no Lewy-related pathology.

neuroscience↗

Single-cell atlas of the human immune system reveals sex-specific dynamics of immunosenescence

Immunosenescence, or immune aging, is characterized by both changes in cell type abundance and a decline in cellular function, leading to increased susceptibility to immune-related diseases. Yet, the extent to which sex influences the dynamic composition of immune cells during immunosenescence remains unknown. Here, we use single-cell RNA sequencing in peripheral blood mononuclear cells from 982 donors to uncover the sex-specific immune aging dynamics. We reveal that aging drives sexually dimorphic compositional and transcriptional shifts, with females showing stronger immune remodeling. Female-specific shifts include the expansion of three cytotoxic CD8+ T effector memory subpopulations and inflammatory monocytes. In addition, female CD4+ central memory T cells exhibit abundance shifts in subpopulations involved in autoimmunity alongside age-associated transcriptional signatures enriched for autoimmune-related pathways. Conversely, males show an age-related expansion of a B cell subpopulation associated with an asymptomatic precursor state to chronic lymphocytic leukemia. These sex-biased, functionally distinct immune subpopulations represent sex-specific hallmarks of immune aging. Our work underscores the complexity and sexual dimorphism of immunosenescence and supports the development of sex-specific strategies to promote healthy aging. Highlights- A single-cell transcriptomic atlas of >1 million PBMCs from 982 donors uncovers sex-specific immune aging trajectories. - Aging females show greater cellular remodeling and stronger transcriptional shifts than males. - Female-specific expansions with age of cytotoxic CD8+ T effector memory cells, inflammatory monocytes, and CD4+ T helper 22 cells reflect a systemic shift toward a pro-inflammatory, self-reactive immune state. - Male-specific B cell shifts reveal expansion of CD5+ clonal subpopulations linked to leukemic precursor states. Graphical Abstract O_FIG O_LINKSMALLFIG WIDTH=200 HEIGHT=158 SRC="FIGDIR/small/622096v2_ufig1.gif" ALT="Figure 1"> View larger version (39K): org.highwire.dtl.DTLVardef@161b6b1org.highwire.dtl.DTLVardef@214d55org.highwire.dtl.DTLVardef@5c3ee8org.highwire.dtl.DTLVardef@263227_HPS_FORMAT_FIGEXP M_FIG C_FIG

bioinformatics↗

The regulatory grammar of human promoters uncovered by MPRA-trained deep learning

One of the major challenges in genomics is to build computational models that accurately predict genome-wide gene expression from the sequences of regulatory elements. At the heart of gene regulation are promoters, yet their regulatory logic is still incompletely understood. Here, we report PARM, a cell-type specific deep learning model trained on specially designed massively parallel reporter assays that query human promoter sequences. PARM requires [~]1,000 times less computational power than state-of-the-art technology, and reliably predicts autonomous promoter activity throughout the genome from DNA sequence alone, in multiple cell types. PARM can even design purely synthetic strong promoters. We leveraged PARM to systematically identify binding sites of transcription factors (TFs) that are likely to contribute to the activity of each natural human promoter. We uncovered and experimentally confirmed striking positional preferences of TFs that differ between activating and repressive regulatory functions, as well as a complex grammar of motif-motif interactions. For example, many, but not all, TFs act as repressors when their binding motif is located near or just downstream of the transcription start site. Our approach lays the foundation towards a deep understanding of the regulation of human promoters by TFs. HighlightsO_LICausality-trained deep learning model PARM captures regulatory grammar of human promoters C_LIO_LIPARM is highly economical, both experimentally and computationally C_LIO_LITranscription factors have different preferred positions for their regulatory activity C_LIO_LIMany (but not all) transcription factors act as repressors when binding downstream of transcription start sites C_LI

genetics↗

Gene expression and eQTL analysis reflect the heterogeneity in the inflammatory status of the duodenal epithelial lining in coeliac disease

In coeliac disease (CeD), the epithelial lining (EL) of the small intestine is severely damaged through a complex auto-inflammatory response that results in intraepithelial lymphocytes (IELs) attacking epithelial cells (ECs). To better understand the changes occurring in the EL in CeD, including after initiation of a gluten-free diet, and the regulatory mechanisms that affect mucosal homeostasis, we investigated ECs and IELs in the CeD duodenal EL using RNA-seq and eQTL analysis on predicted cell types. The study included 82 duodenal biopsies from volunteers, grouped into controls (CTRL), gluten-free diet treated CeD (TCD) and untreated CeD (UCD). We identified 2,868 differential expressed genes, which clustered into four sets with specific functions related to CeD. Two sets, one upregulated for cell cycle function and one downregulated for digestion, transmembrane transport, and laminin pathways, defined three groups of samples based on inflammation status: non-inflamed, mild inflammation or severe inflammation. These inflammation states correlated with but were not specific to disease state. The remaining two sets of genes were enriched for immune, extracellular matrix, and barrier functions. These latter two sets allowed the classification of samples into their disease conditions: CTRL, TCD, and UCD. Finally, deconvoluting eQTL effects from ECs and immune cells in the EL identified 7 and 9 cell-type-mediated eQTL genes, respectively. In sum, we identified genes expressed in the duodenal EL whose expression might be used as biomarkers to assess CeD condition and its mucosal and immune status. HighlightsO_LIGene expression in epithelial lining in CeD patients reflects inflammation status C_LIO_LIThe transcriptome can classify epithelium as no, mild, or severe inflammation C_LIO_LICell cycle, absorption, and basal lamina genes are indicators of mucosal damage C_LIO_LIImmune and extracellular matrix genes distinguish untreated from treated cases C_LIO_LIPredicted-cell-type eQTLs pinpoint effects in immune and epithelial cells C_LI

immunology↗

An iPSC-derived small intestine-on-chip with self-organizing epithelial, mesenchymal and neural cells

Human induced pluripotent stem cell (hiPSC)-derived intestinal organoids are valuable tools for researching developmental biology and personalized therapies, but their closed topology and relative immature state limits their applications. Here we use organ-on-chip technology to develop a hiPSC-derived intestinal barrier with apical and basolateral access in a more physiological in vitro microenvironment. To replicate growth factor gradients along the crypt- villus axis, we locally exposed the cells to expansion and differentiation media. In these conditions, intestinal epithelial cells self-organize into villus-like folds with physiological barrier integrity and myofibroblast and neural subtypes emerge and form a layer in the bottom channel underneath the epithelial tissue. The growth factor gradients efficiently balance dividing and mature cell types and induce an intestinal epithelial composition, including absorptive and secretory lineages, resembling the composition of the human adult small intestine. The result is a well-characterized hiPSC-derived intestine-on-chip system that can facilitate personalized studies on physiological processes and therapy development in the human small intestine.

cell biology↗

Linking the gut microbiome to host DNA methylation by a discovery and replication epigenome-wide association study

Both gene methylation and the gut microbiome are partially determined by host genetics and partially by environment. We investigated the relations between gene methylation in blood and the abundance of common gut bacteria profiled by 16s rRNA gene sequencing in two population-based Dutch cohorts: LifeLines-Deep (LLD, n = 616, discovery) and the Netherlands Twin Register (NTR, n = 296, replication). In LLD, we also explored microbiome composition using data generated by shotgun metagenomic sequencing (n = 683). We then investigated if genetic and environmental factors can explain the methylation-microbiota associations in a set of 78 associated CpG-taxa pairs from the EWAS meta-analysis. In both cohorts, blood and stool samples were collected within 2 weeks of each other. Methylation was profiled in blood samples using the Illumina 450K array. Methylation and microbiome analysis pipelines were harmonized across cohorts. Epigenome-wide association study (EWAS) of microbial features were analysed using linear regression with adjustment for technical covariates. Discovery and replication analysis using 16s data identified two independent CpGs associated with the genus Eggerthella: cg16586104 (Pmeta-analysis = 3.21 x 10-11) and cg12234533 (Pmeta-analysis = 4.29 x 10-10). While we did not find human genetic variants that could explain the associated CpG-taxa/pathway pairs, we show that microbiome can mediate the effect of environmental factors on epigenetics. In this first association study linking epigenome to microbiome, we found and replicated the associations of two CpGs to the abundance of genus Eggerthella and identified microbiome as a mediator of the exposome.

genomics↗

Arabidopsis hydathodes are sites of intense auxin metabolism and nutrient scavenging

Hydathodes are small organs located on the leaf margins of all vascular plants. They release excess xylem sap through guttation when stomata are closed or when the humidity level is high. Many promoter analyses have suggested other hydathode functions in metabolite transport and auxin metabolism, but experimental demonstration is still lacking. Here, we compared the transcriptomic and metabolomic features of mature Arabidopsis hydathodes to the leaf blade. 1460 differentially-expressed genes were identified revealing that genes related to auxin metabolism, transport, stress, DNA, plant cell wall, RNA or wax were on average more expressed in hydathodes. On the other hand, genes involved in glucosinolate metabolism, sulfation pathway, metal handling or photosynthesis were downregulated in hydathodes. In hydathodes, there are an increased expression of auxin transcriptional regulators and biosynthetic genes, a lower expression of auxin transport genes and a differential expression of genes related to its vacuolar storage that is consistent with increased contents of free and conjugated auxin. We also found that ca. 78% of the total content of 52 xylem sap metabolites were removed from guttation fluid at the hydathode level. Using reverse genetics, we showed that the capture of nitrate and phosphate in the guttation fluid relies on the NRT2.1 and PHT1;4 transporters, respectively. Thus, hydathodes absorb a significant part of xylem sap nutrients, limiting the loss of valuable chemicals during guttation. Our transcriptomic and metabolomic analyses reveal an organ with its own transcriptomic and physiological identity and highlight hydathode biological processes that may impact the whole plant. One sentence summaryTranscriptome and physiological analysis of mature and healthy hydathodes of Arabidopsis demonstrates that those organs are sites of intense auxin metabolism and nutrient scavenging

plant biology↗

Unbiased identification of unknown cellular and environmental factors that mediate eQTLs using principal interaction component analysis

Expression quantitative trait loci (eQTL) can reveal the regulatory mechanisms of trait associated variants. eQTLs are highly cell-type and context-specific, but often these contexts are unknown or not measured. Here, we introduce PICALO (Principal Interaction Component Analysis through Likelihood Optimization), an unbiased method to identify known and hidden contexts that influence eQTLs. PICALO uses expectation maximization to identify latent components, referred to as Principal Interaction Components (PIC), that interact with genotypes to maximize explained eQTL effect-sizes. We applied PICALO to bulk RNA-seq eQTL datasets in blood (n=2,932) and brain (n=2,440). We identify 31 PICs in blood, interacting with 4,169 (32%) unique cis-eQTLs (BH-FDR[&le;]0.05). In brain, we identified 21 PICs, interacting with 4,058 (39%) unique cis-eQTLs (BH-FDR[&le;]0.05). These PICs are associated with RNA quality, cell type composition or environmental influences. Furthermore, PICs clearly disentangle distinct eQTL contexts, for example technical from non-technical factors. Combined, 3,065 unique genes showed a cis-eQTL effect that is dependent on a cell type or other non-technical context, emphasizing the value of methods like PICALO. PICALO is robust, works well with heterogeneous datasets, yields reproducible interaction components, and identifies eQTL interactions and contexts that would have been missed when using cell counts or expression based principal components. Since PICALO allows for the identification of many context-dependent eQTLs without any prior knowledge of such contexts, this method can help to reveal and quantify the influence of previously unknown environmental factors that play a role in common diseases.

genomics↗

Integration of Candida albicans-induced single-cell gene expression data and circulatory protein concentrations reveal genetic regulators of inflammation

Both gene expression and protein concentrations are regulated by genetic variants. Exploring the regulation of both eQTLs and pQTLs simultaneously in a context- and cell-type dependent manner may help to unravel mechanistic basis for genetic regulation of pQTLs. Here, we performed meta-analysis of Candida albicans-induced pQTLs from two population-based cohorts and intersected the results with Candida-induced cell-type specific expression association data (eQTL). This revealed systematic differences between the pQTLs and eQTL, with only 35% of common genetic variants modulating both intermediate molecular phenotypes. By taking advantage of the tightly co-regulated pattern of the proteins, we also identified SNPs affecting protein network upon Candida stimulations. Colocalization of pQTLs and eQTLs signals implicated several genomic loci including MMP-1 and AMZ1. Analysis of Candida-induced single cell gene expression data implicated specific cell types that exhibit significant expression QTLs upon stimulation. By highlighting the role of trans-regulatory networks in determining the abundance of proteins in blood, our study serve as a framework to gain insights into the mechanisms of genetic regulation of protein levels in a context-dependent manner.

genomics↗

Identification of genetic variants that impact gene co-expression relationships using large-scale single-cell data

BackgroundExpression quantitative trait loci (eQTL) studies have shown how genetic variants affect downstream gene expression. To identify the upstream regulatory processes, single-cell data can be used. Single-cell data also offers the unique opportunity to reconstruct personalized co-expression networks--by exploiting the large number of cells per individual, we can identify SNPs that alter co-expression patterns (co-expression QTLs, co-eQTLs) using a limited number of individuals. ResultsTo tackle the large multiple testing burden associated with a genome-wide analysis (i.e. the need to assess all combinations of SNPs and gene pairs), we conducted a co-eQTL meta-analysis across four scRNA-seq peripheral blood mononuclear cell datasets from three studies (reflecting 173 unique participants and 1 million cells) using a novel filtering strategy followed by a permutation-based approach. Before analysis, we evaluated the co-expression patterns to be used for co-eQTL identification using different external resources. The subsequent analysis identified a robust set of cell-type-specific co-eQTLs for 72 independent SNPs that affect 946 gene pairs, which we then replicated in a large bulk cohort. These co-eQTLs provide novel insights into how disease-associated variants alter regulatory networks. For instance, one co-eQTL SNP, rs1131017, that is associated with several autoimmune diseases affects the co-expression of RPS26 with other ribosomal genes. Interestingly, specifically in T cells, the SNP additionally affects co-expression of RPS26 and a group of genes associated with T cell-activation and autoimmune disease. Among these genes, we identified enrichment for targets of five T-cell-activation-related transcriptional factors whose binding sites harbor rs1131017. This reveals a previously overlooked process and pinpoints potential regulators that could explain the association of rs1131017 with autoimmune diseases. ConclusionOur co-eQTL results highlight the importance of studying gene regulation at the context-specific level to understand the biological implications of genetic variation. With the expected growth of sc-eQTL datasets, our strategy--combined with our technical guidelines--will soon identify many more co-eQTLs, further helping to elucidate unknown disease mechanisms.

genetics↗

OTTERS: A powerful TWAS framework leveraging summary-level reference data

Most existing TWAS tools require individual-level eQTL reference data and thus are not applicable to summary-level reference eQTL datasets. The development of TWAS methods that can harness summary-level reference data is valuable to enable TWAS in broader settings and enhance power due to increased reference sample size. Thus, we develop a TWAS framework called OTTERS (Omnibus Transcriptome Test using Expression Reference Summary data) that adapts multiple polygenic risk score (PRS) methods to estimate eQTL weights from summary-level eQTL reference data and conducts an omnibus TWAS. We show that OTTERS is a practical and powerful TWAS tool by both simulations and application studies.

genetics↗

Demuxafy: Improvement in droplet assignment by integrating multiple single-cell demultiplexing and doublet detection methods

Recent innovations in droplet-based single-cell RNA-sequencing (scRNA-seq) have provided the technology necessary to investigate biological questions at cellular resolution. With the ability to assay thousands of cells in a single capture, pooling cells from multiple individuals has become a common strategy. Droplets can subsequently be assigned to a specific individual by leveraging their inherent genetic differences, and numerous computational methods have been developed to address this problem. However, another challenge implicit with droplet-based scRNA-seq is the occurrence of doublets - droplets containing two or more cells. The inaccurate assignment of cells to individuals or failure to remove doublets contribute unwanted noise to the data and result in erroneous scientific conclusions. Therefore, it is essential to assign cells to individuals and remove doublets accurately. We present a new framework to improve individual singlet classification and doublet removal through a multi-method intersectional approach. We developed a framework to evaluate the enhancement in donor assignment and doublet removal through the consensus intersection of multiple demultiplexing and doublet detecting methods. The accuracy was assessed using scRNA-seq data of [~]1.4 million peripheral blood mononucleated cells from 1,034 unrelated individuals and [~]90,000 fibroblast cells from 81 unrelated individuals. We show that our approach significantly improves droplet assignment by separating singlets from doublets and classifying the correct individual compared to any single method. We show that the best combination of techniques varies under different biological and experimental conditions, and we present a framework to optimise cell assignment for a given experiment. We offer Demuxafy (https://demultiplexing-doublet-detecting-docs.readthedocs.io/en/latest/index.html) - a framework built-in Singularity to provide clear, consistent documentation of each method and additional tools to simplify and improve demultiplexing and doublet removal. Our results indicate that leveraging multiple demultiplexing and doublet detecting methods improves accuracy and, consequently, downstream analyses in multiplexed scRNA-seq experiments.

genomics↗

Omics-informed CNV calls reduce false positive rate and improve power for CNV-trait associations

Copy number variations (CNV) are believed to play an important role in a wide range of complex traits but discovering such associations remains challenging. Whilst whole genome sequencing (WGS) is the gold standard approach for CNV detection, there are several orders of magnitude more samples with available genotyping microarray data. Such array data can be exploited for CNV detection using dedicated software (e.g., PennCNV), however these calls suffer from elevated false positive and negative rates. In this study, we developed a CNV quality score that weights PennCNV calls (pCNV) based on their likelihood of being true positive. First, we established a measure of pCNV reliability by leveraging evidence from multiple omics data (WGS, transcriptomics and methylomics) obtained from the same samples. Next, we built a predictor of omics-confirmed pCNVs, termed omics-informed quality score (OQS), using only PennCNV software output parameters. Promisingly, OQS assigned to pCNVs detected in close family members was up to 35% higher than the OQS of pCNVs not carried by other relatives (P < 3.0-10-90), outperforming other scores. Finally, in an association study of four anthropometric traits in 89,516 Estonian Biobank samples, the use of OQS led to a relative increase in the trait variance explained by CNVs of up to 34% compared to raw pCNVs or previous quality scores. Overall, we put forward a flexible framework to improve any CNV detection method leveraging multi-omics evidence, applied it to improve PennCNV calls and demonstrated its utility by improving the statistical power for downstream association analyses.

bioinformatics↗

Integration of public DNA methylation and expression networks via eQTMs improves prediction of functional gene-gene associations

Gene co-expression networks can be used to infer functional relationships between genes, but they do not work well for all genes. We investigated whether DNA methylation can provide complementary information for such genes. We first carried out an eQTM meta-analysis of 3,574 gene expression and methylation samples from blood, brain and nasal epithelial brushed cells to identify links between methylated CpG sites and genes. This revealed 6,067 significant eQTM genes, and we observed that histone modification information is predictive of both eQTM direction and presence, enabling us to link many CpG sites to genes. We then generated a co-methylation network - MethylationNetwork - using 27,720 publicly available methylation profiles and integrated it with a public RNA-seq co-expression dataset of 31,499 samples. Here, we observed that MethylationNetwork can identify experimentally validated interacting pairs of genes that could not be identified in the RNA-seq datasets. We then developed a novel integration pipeline based on CCA and used the integrated methylation and gene networks to predict gene pairs reported in the STRING database. The integrated network showed significantly improved prediction performance compared to using a DNA co-methylation or a gene co-expression network alone. This is the first study to integrate data from two -omics layers from unmatched public samples across different tissues and diseases, and our results highlight the issues and potential of integrating public datasets from multiple molecular phenotypes. The eQTMs we identified can be used as an annotation resource for epigenome-wide association, and we believe that our integration pipeline can be used as a framework for future -omics integration analyses of public datasets. We provide supporting materials and results, including the harmonized DNA methylation data from multiple tissues and diseases in https://data.harmjanwestra.nl/comethylation/, the discovered and predicted eQTMs, the corresponding CCA components and the trained prediction models in a Zenodo repository (https://zenodo.org/record/4666994). We provide notebooks to facilitate use of the proposed pipeline in a GitHub repository (https://github.com/molgenis/methylationnetwork).

bioinformatics↗

Genetic, parental and lifestyle factors influence telomere length

The average length of telomere repeats (TL) declines with age and is considered to be a marker of biological ageing. Here, we measured TL in six blood cell types from 1,046 individuals using the clinically validated Flow-FISH method. We identified remarkable cell-type-specific variations in TL. Host genetics, environmental, parental and intrinsic factors such as sex, parental age, and smoking are associated to variations in TL. By analysing the genome-wide methylation patterns, we identified that the association of maternal, but not paternal, age to TL is mediated by epigenetics. Coupling these measurements to single-cell RNA-sequencing data for 62 participants revealed differential gene expression in T-cells. Genes negatively associated with TL were enriched for pathways related to translation and nonsense-mediated decay. Altogether, this study addresses cell-type-specific differences in telomere biology and its relation to cell-type-specific gene expression and highlights how perinatal factors play a role in determining TL, on top of genetics and lifestyle.

cell biology↗