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Galbraith, M.

Publications and source records attributed to Galbraith, M..

3 recordsLinked to original sources

Attenuated estrogen signaling disrupts placentation and drives trophoblast defects in Down syndrome

Proper establishment of the primitive placenta and subsequent tissue homeostasis in the mature placenta are critical for successful pregnancy in humans. Placental insufficiency is associated with adverse pregnancy outcomes, including fetal growth restriction, preeclampsia, and pregnancy loss. Moreover, emerging evidence suggests that placental defects are associated with long-term health challenges that manifest well into adulthood; yet the etiologies of such diseases are largely unknown. Defining the mechanistic basis for placental deficiencies, therefore, has important implications for improving both reproductive health and the lifelong well-being of affected children. Down syndrome is characterized by placental defects of unknown mechanistic origin, and notably, individuals with Down syndrome are at increased risk of developing diseases commonly associated with placental insufficiency later in life. Using induced pluripotent stem cells from Down syndrome patients, we found that stem cell-based embryo models (i.e., blastoids) and directed differentiation systems recapitulate trophoblast cell fate defects observed in placentas affected by Down syndrome. Furthermore, we demonstrate that attenuated estrogen signaling contributes to placental syncytialization defects and identify NRIP1, a transcriptional corepressor of estrogen receptor that is located on chromosome 21, as a key driver of trophoblast cell fate defects. Increased gene dosage of NRIP1 in an otherwise diploid cell line phenocopies cell fate defects observed in trophoblasts affected by Down syndrome. Our study suggests that estrogen signaling is a crucial regulator of trophoblast development and may serve as a potential target for therapeutic intervention. Highlights and eTOC blurbO_LIEstrogen signaling mediates syncytiotrophoblast fusion C_LIO_LIHuman iPS cells provide a tractable model for trophoblast defects in Down syndrome C_LIO_LITrophoblast differentiation and estrogen signaling are disrupted in Down syndrome C_LIO_LIIncreased NRIP1 expression is sufficient to induce trophoblast defects C_LI Logsdon and colleagues apply patient-derived induced pluripotent stem cells to recapitulate placentation defects observed in Down syndrome. The authors demonstrate that attenuated estrogen signaling disrupts trophoblast differentiation and identify NRIP1, a gene found on chromosome 21 that dampens estrogen signaling, as a regulator of trophoblast maturation. NRIP1 and estrogen signaling may represent important therapeutic targets for infertility and Down syndrome.

developmental biology↗

Benchmarking strain-level profiling of Escherichia coli in short-read gut metagenomes

2.Metagenomes offer the potential to characterise Escherichia coli strain-level diversity within the human gut microbiome, informing our understanding of colonisation diversity and the genetic features distinguishing infection from carriage. Among numerous reference-based tools for short-read metagenomic strain-level profiling, the best approach remains unclear. Here, we benchmarked six published tools--PanTax, PathoScope, StrainGE, Strainify, StrainR2 and StrainScan--for their ability to detect co-existing strains of E. coli and estimate their relative abundance across real and simulated metagenomes of increasing complexity with varying reference database composition. In the ZymoBIOMICS(R) D6331 dataset, only PanTax achieved zero error when predicting the equal abundance of five E. coli strains. In a differentially abundant four-strain mock community dataset (SRR13355226), StrainScan had the lowest mean absolute proportional error (0.89), driven by reduced sensitivity (0.5), followed by PathoScope (4.08). Across simulated metagenomes reflecting the healthy adult gut microbiome, all tools demonstrated high sensitivity ([≥]0.833), but specificity, precision and F1 score were selectively improved in some tools through detection thresholds to remove low abundance false positives. Outright, StrainGE achieved the highest F1 score (0.978). Predicted relative abundances of the E. coli K12-MG1655 (phylogroup A) and O157:H7 Sakai (phylogroup E) strains spiked into simulated metagenomes across varying abundance ratios were generally accurate, with PanTax and StrainR2 showing the lowest mean absolute proportional error (0.06). When truly present strains were removed from the reference database, out-of-phylogroup assignments were observed for some tools. Collectively, our results demonstrate that published metagenomic strain-level profiling tools vary in their ability to profile E. coli strains, indicating that method selection should be guided by intended application. These findings will facilitate characterisation of E. coli strain-level diversity within short-read gut metagenomes with greater accuracy than previously possible. 3. Impact statementStrain-level diversity within the human gut microbiome can be important for human health, with species such as Escherichia coli existing as both commensal and pathogenic strains. Most existing gut microbiome datasets are from short-read i.e., Illumina, sequencing, and numerous bioinformatic tools have been developed to profile strain-level variation from these data. However, the existing literature is often difficult to navigate given that the available tools have been benchmarked in various ways and are subject to author bias. This is, to our knowledge, the first independent benchmarking of six published tools for profiling E. coli at strain-level resolution from short-read metagenomes. Using both real and simulated datasets of increasing complexity, we demonstrate substantial variation in tool performance in terms of strain detection and relative abundance estimation, highlighting that tool choice should be guided by the specific research question, as no single method performs optimally across all scenarios. This work provides an unbiased framework for tool selection and will support more accurate and reproducible E. coli strain-level analyses in gut microbiome research from short-metagenomic data. 4. Data summaryThe authors confirm all supporting data, code and protocols have been provided within the article or through supplementary data files. Supplementary methods, six supplementary tables and four supplementary figures are available in the online Supplementary Material. Code for simulating metagenomes using InSilicoSeq, SLURM job scripts for the simulated metagenomes dataset and R visualization and statistical analysis scripts are available within a dedicated public GitHub repository (https://github.com/mattgal11/benchmarking_short_read_strain_profilers). The following supplementary data are available on FigShare (https://doi.org/10.6084/m9.figshare.32125474): O_LINormalised per-contig relative abundances for 98 species assemblies used to construct the baseline gut microbiome profile for InSilicoSeq metagenome simulation (Normalised_relative_abundance_for_InSilicoSeq_simulated_metagenomes_ gut_microbiome_profile.csv) C_LIO_LIZymoBIOMICS(R) D6331 gut microbiome standard dataset predicted relative abundance data (Zymobiomics_D6331_raw_predicted_abundance.csv) C_LIO_LISRR13355226 mock community (99% human reads; 1% E. coli reads) paired-end reads with human reads depleted (SRR13355226_depleted_R1.fastq.gz & SRR13355226_depleted_R2.fastq.gz) C_LIO_LISRR13355226 mock community dataset raw predicted abundance data, with and without human read removal (SRR13355226_raw_predicted_abundance_with_and_without_human_read_r emoval.csv) C_LIO_LISimulated metagenomes dataset raw call types and detection metric values with increasing detection thresholds (Simulated_metagenomes_raw_call_type_assingments_and_detection_thres holds.csv) C_LIO_LISimulated metagenomes dataset (all references) predicted relative abundance data (Simulated_metagenomes_all_references_raw_predicted_abundances.csv) C_LIO_LISimulated metagenomes dataset (all references) mapped reads for PathoScope and Strainify (all_refs_pathoscope_reads_mapped.csv & all_refs_strainify_reads_mapped.csv) C_LIO_LISimulated metagenomes dataset (reduced reference database) predicted relative abundance data (Simulated_metagenomes_K12_and_Sakai_removed_from_reference_datab ase_raw_predicted_abundance.csv) C_LI

bioinformatics↗

Comprehensive Longitudinal ctDNA Monitoring in Metastatic Cancer Patients Treated with an Individualized Neoantigen-directed Vaccine

PurposeCirculating-tumor DNA (ctDNA) is an emerging, minimally invasive diagnostic and prognostic biomarker for patients receiving a variety of cancer therapies. Comprehensive and robust longitudinal monitoring of ctDNA can provide an understanding of tumor burden, heterogeneity, and response or resistance to treatment. Experimental DesignctDNA of 28 metastatic cancer patients receiving an individualized neoantigen-directed immunotherapy was monitored longitudinally, up to two years, using a unique hybrid next generation sequencing assay targeting tumor-informed and tumor-naive variants. Patient-specific panels were designed targeting an average of 144 variants per patient. A tumor-naive universal panel was also designed for inclusion with patient-specific panels to monitor recurrently mutated tumor hotspots (e.g., KRAS and TP53) and genes implicated in immunotherapy resistance (B2M, TAP1/2). ResultsAnalytical characterization of the assay established linearity with a mean variant allele frequency (VAF) [≥]0.049%, and a variant-level limit of detection (LOD95) of 0.12%. Tumor-informed variants were detected in 26/28 patients, and de novo variants were observed in 25/28 patients. HLA LOH was also observed. Longitudinal ctDNA data provided key insights into patients responses to vaccine treatment. ConclusionsThe hybrid design of the ctDNA monitoring assay provides the sensitivity and specificity required for evaluating patient samples undergoing individualized therapy. It provides an improved capability to understand patient response to experimental therapies and further supports the utility of ctDNA as a cancer biomarker.

genomics↗