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Biology subjects

Morimoto, M.

Publications and source records attributed to Morimoto, M..

At least 19 recordsLinked to original sources

A plasma metabolomics workflow for breast cancer detection using quantitative GC/MS and machine learning

Blood-based metabolomic profiling has been widely investigated for breast cancer (BC) detection; however, clinical implementation remains limited due to variability in sample handling, analytical reproducibility, and overfitting during statistical analysis. We established a plasma GC/MS metabolomics workflow for discriminating BC from healthy controls (HC) using conventional machine-learning algorithms. Plasma samples (n = 360; BC = 180, HC = 180) were collected prospectively under standardized preanalytical conditions before surgery and the initiation of systematic anticancer therapy and analyzed using a quantitative GC/MS platform with automated derivatization. Feature selection and model development were conducted using three machine-learning (ML) algorithms (Lasso logistic regression (LR), random forest classifier (RFC), and support vector machine (SVM)). A total of 45 metabolite candidate biomarkers were identified, and the optimal number of metabolite features for each algorithm was estimated by a recursive feature elimination (RFE)-based strategy. The best-performing models achieved area under the ROC curve values (AUC) of 0.910 (LR), 0.893 (RFC), and 0.843 (SVM). We selected prioritizing candidate biomarkers consistently expressed across the multi-algorithm pipeline. A bagging ensemble model improved stability (AUC = 0.911) and reduced false-positive predictions in the independent HC dataset. In addition, model stability with respect to false-positive predictions was assessed using an independent HC cohort (n = 15) that was collected at a separate institution. These results indicate that a plasma metabolomics workflow combined with conventional multi-algorithm ML, algorithm-specific feature selection, and independent assessment provides stable discrimination between BC and HC in a moderately sized cohort.

cancer biology↗

Ductal myofibroblasts reactivate contractile program to stabilize alveolar architecture during lung regeneration

The alveolar sac architecture is essential for efficient gas exchange and must be precisely maintained throughout life; however, how this delicate structure is preserved during adult regeneration remains poorly understood. Using a mouse pneumonectomy model, we found that Lgr6+ Hhip+ ductal myofibroblasts, a poorly characterized mesenchymal population, are indispensable for maintaining alveolar integrity during lung regrowth. Comprehensive characterization using single-cell transcriptomics, mouse genetics, and pharmacological assays demonstrated that these ductal myofibroblasts secrete myogenic factors, most notably CCN4, to reactivate a myogenic program that converts them into contractile PA-DMFs, thereby preserving alveolar architecture. Lineage-tracing further revealed that these ductal myofibroblasts originate from embryonic MCAM- SMA+ distal progenitors via subepithelial TGF-{beta} signaling, serving as a lifelong guardian of alveolar structural integrity. Notably, cross-species analysis identified an analogous population of LGR6+ fibromyocytes in human respiratory bronchioles. Together, these findings indicate ductal myofibroblasts as a developmentally programmed cell population that reactivate a contractile program to structurally support the regeneration of adult lungs.

developmental biology↗

Environment-specific mechanosensing preserves a common bleb-based migratory program across diverse embryonic environments

Cell migration frequently requires cells to traverse tissue environments with distinct physical and biochemical properties. How migrating cells preserve a common migratory program while adapting to these heterogeneous environments remains poorly understood. Here, we show that chick primordial germ cells (PGCs) preserve a common bleb-based migratory mode throughout embryogenesis despite migrating through mechanically distinct tissues. PGCs formed membrane blebs during both intravascular crawling and migration through the dorsal mesentery. However, nuclear envelope (NE) unfolding and activation of the NE-cPLA2 pathway occurred specifically during migration through the mechanically confined dorsal mesentery, where this pathway was required for bleb formation and efficient migration. In contrast, bleb formation during vascular crawling occurred independently of the NE-cPLA2 pathway, demonstrating that distinct molecular mechanisms can generate the same migratory behavior in different tissue environments. Together, these findings suggest that diverse environmental cues converge on a conserved bleb-forming machinery. We propose a hierarchical model in which migrating cells preserve a common migratory program by flexibly switching the upstream molecular mechanisms that initiate bleb formation according to the tissue environment.

developmental biology↗

Bile acid chemosensation in mammals supports species and gut microbiome evaluation

The rodent vomeronasal system, also known as the accessory olfactory system (AOS), detects excreted chemosignals that guide social, reproductive, and defensive behaviors. Excretions sensed by the AOS contain blends of organic chemicals that reflect the biological identity of the emitter, but it remains unclear how the AOS extracts useful information from these complex natural cues. Vomeronasal sensory neurons (VSNs) sense bile acids, a diverse class of molecules excreted by vertebrates in feces. Using mass spectrometry, we found that patterns of bile acid excretion - not unique molecules - provide sufficient information to decode the emitters species, diet, and gut microbiome status. Using population calcium imaging, we found that VSN populations demonstrate an inverse relationship between natural bile acid abundance and population response magnitude and support efficient information encoding through stimulus "whitening." VSNs showed maximum sensitivity to taurine-conjugated bile acids, a novel class of vomeronasal ligands found at low natural abundance levels, that have high theoretical discriminatory value. VSN tuning patterns aligned strongly with theoretical axes for discriminating reptilian predators from vegetarians, and between mice with different gut microbiome states. These results reveal that the AOS extracts biological state from bile acid emission patterns through enhanced sensitivity to rare, information-rich ligands.

neuroscience↗

Bleb-based extravasation: conserved morphodynamics, divergent calcium control

Extravasation, the exit of cells from blood vessels, is essential in both development and disease, from germ cell migration to cancer metastasis. We show that all extravasating cells examined, avian primordial germ cells and diverse human cancer lines, form Ca{superscript 2}-dependent membrane blebs during transendothelial migration. Yet the source of Ca{superscript 2} diverges by lineage. PGCs and HT-1080 fibrosarcoma cells rely on store-operated Ca{superscript 2} entry (SOCE), whereas epithelial cancer cells such as PC-3 and MDA-MB-231 use IPR-mediated Ca{superscript 2} release from the endoplasmic reticulum. These findings establish bleb-based extravasation as a conserved morphodynamic strategy powered by distinct regulatory modules. Evolutionarily, they map onto an ancestral ER-release pathway and a metazoan-derived SOCE pathway. Conceptually, cancer blebbing emerges as a composite strategy that reactivates both ancient survival programs and developmental toolkits to maximize invasive success. This unified framework highlights Ca{superscript 2} supply as a critical bottleneck of vascular escape, offering new angles for targeting metastatic dissemination.

cell biology↗

Primitive Hepatoblasts Driving Early Liver Development

The embryonic development of the liver is initiated by the emergence of hepatoblasts, originating from the ventral foregut endoderm adjacent to the heart. Here, we identify and characterize a previously unrecognized population of early hepatoblasts at the ventroposterior part of the emerging liver bud, traced from Cdx2-positive endoderm progenitors, which we term primitive hepatoblasts. Mouse and human single-cell transcriptomics reveals the expression of both canonical hepatoblast markers TBX3, FGB, and KRT8/18 and primitive-specific mesenchymal markers ID3, VIM, and GATA4. Lineage tracing revealed the notable contribution up to 12.6% of LIV2+ hepatoblasts at E11.5 but diminishes in late fetal and postnatal development. Epigenetic and functional perturbation studies further uncover that primitive hepatoblast emergence is primed by WNT-suppression on RA-permissive CDX2+FOXA2+ progenitors. Furthermore, human pluripotent stem cell-derived primitive hepatoblast-like cells secrete pleiotrophin and midkine to amplify hepatoblast populations and develop epithelial-mesenchymal hybrid tissues in vivo. Our results provide a new framework for understanding lineage heterogeneity during early hepatogenesis and offer revised insights into strategies to model normal and abnormal liver development.

developmental biology↗

Mammalian chemosensory bile acid detection supports species and gut microbiome evaluation

Long abstractThe rodent accessory olfactory system (AOS) detects environmental chemosignals and guides social and survival-oriented behaviors. Fecal bile acids activate neurons in the AOS, potentially serving as mammalian pheromones and kairomones, but few molecules in this large class have been evaluated thus far. We used live volumetric Ca2+ imaging to screen naturally occurring bile acids for their capacity to activate peripheral vomeronasal sensory neurons (VSNs). We found that taurine-conjugated bile acids (tauro-BAs), including taurine-conjugates of cholic acid, deoxycholic acid, lithocholic acid, and chenodeoxycholic acid (TCA, TDCA, TLCA, TCDCA, respectively) activate large populations of VSNs at sub-micromolar concentrations. Tauro-BA-sensitive VSNs rarely responded to unconjugated (CA, CDCA, DCA, LCA), glycine-conjugated (GCA, GDCA, GLCA, GCDCA), or keto-conjugated (7-keto DCA, 12-keto DCA, 7-keto LCA) bile acids. Tauro-BA-sensitive VSNs were also insensitive to well-studied sulfated steroids, suggesting tauro-BAs activate vomeronasal receptors that have not yet been de-orphaned. Among the tauro-BAs, TDCA displayed particularly strong potency, activating many VSNs at sub-micromolar concentrations. Tauro-BAs were not detectable in mouse fecal extracts by mass spectrometry, but were found in reptile fecal extracts and germ-free mouse fecal extracts. VSN responses to germ-free and conventional mouse fecal extracts, conjugated bile acids, and tauro-BAs revealed that non-overlapping populations of VSNs respond to germ-free and conventional mouse feces. A subset of VSNs that were activated by germ-free mouse fecal extracts responded to tauro-BAs, whereas VSNs responsive to conventionally fecal extracts responded to unconjugated bile acids. In vivo exposure to TDCA alone, and to mouse fecal extracts spiked with TDCA, elicited mild aversion and stress-associated behaviors in a non-social context (avoidance, digging, grooming, etc.). These studies establish tauro-BAs as a novel class of aversive vomeronasal ligands that vary in feces across species and gut microbiomes. Short abstractThe rodent accessory olfactory system (AOS) detects environmental chemosignals and guides social and survival-oriented behaviors. Fecal bile acids activate neurons in the AOS, but the full repertoire of bile acids that activate VSNs is not known. A live Ca2+ imaging screen revealed that VSNs were highly activated by taurine-conjugated bile acids (tauro-BAs) at sub-micromolar concentrations. Tauro-BAs were undetectable in conventionally raised mouse fecal extracts, but were found in reptile fecal extracts and germ-free mouse fecal extracts. VSN activity to tauro-BAs overlapped that of germ-free mouse fecal extracts, suggesting a link between bile acid chemosensation and the gut microbiome. When mice were exposed to taurodeoxycholic acid in vivo, they displayed mild aversion and stress-associated behaviors.

neuroscience↗

Modeling epithelial homeostasis and perturbation in three-dimensional human esophageal organoids

BackgroundEsophageal organoids from a variety of pathologies including cancer are grown in Advanced Dulbeccos Modified Eagle Medium-Nutrient Mixture F12 (hereafter ADF). However, the currently available ADF-based formulations are suboptimal for normal human esophageal organoids, limiting the ability to compare normal esophageal organoids with those representing a given disease state. MethodsWe have utilized immortalized normal human esophageal epithelial cell (keratinocyte) lines EPC1 and EPC2 and endoscopic normal esophageal biopsies to generate three-dimensional (3D) organoids. To optimize ADF-based medium, we evaluated the requirement of exogenous epidermal growth factor (EGF) and inhibition of transforming growth factor-(TGF)-{beta} receptor-mediated signaling, both key regulators of proliferation of human esophageal keratinocytes. We have modeled human esophageal epithelial pathology by stimulating esophageal 3D organoids with interleukin (IL)-13, an inflammatory cytokine, or UAB30, a novel pharmacological activator of retinoic acid signaling. ResultsThe formation of normal human esophageal 3D organoids was limited by excessive EGF and intrinsic TGF{beta} receptor-mediated signaling. In optimized HOME0, normal human esophageal organoid formation was improved, whereas IL-13 and UAB30 induced epithelial changes reminiscent of basal cell hyperplasia, a common histopathologic feature in broad esophageal disease conditions including eosinophilic esophagitis. Conclusions: HOME0 allows modeling of the homeostatic differentiation gradient and perturbation of the human esophageal epithelium while permitting a comparison of organoids from mice and other organs grown in ADF-based media.

cell biology↗

The yolk sac vasculature in early avian embryo provides a novel model for the analysis of cancer extravasation

Hematogenous metastasis, a key trait of cancer cells, involves a complex sequence of cell migration steps, including intravasation, circulation, arrest in capillary vessels, and extravasation. Among these steps, extravasation is challenging to image in amniotes like humans and mice due to its unpredictable timing and location, limiting our understanding of cellular and molecular mechanisms through imaging. Establishing a new cancer carrier model with high-resolution imaging capabilities in amniotes is crucial. In this study, we investigated the yolk sac vasculature (YSV) of early avian embryos (chickens and quail) as a new model for studying extravasation, offering excellent imaging capabilities. We examined the YSV structure and attempted fluorescent labeling to enhance visibility. We then injected mCherry-labeled HT-1080 cells into YSV and observed their behavior, revealing distinct morphologies and extravasation dynamics. Our findings suggest that the YSV model holds promise as a novel cancer carrier model for elucidating cellular and molecular mechanisms through imaging-based approaches.

cancer biology↗

Tumor Immune Cell Targeting Chimeras (TICTACs) For Targeted Depletion of Macrophage-Associated Checkpoint Receptors

Immune cells in the tumor microenvironment are not only powerful regulators of immunosuppression and tumorigenesis, but also a dominant cell population, with tumor-associated macrophages (TAMs) comprising up to 50% of solid tumor mass. Immunotherapies such as immune checkpoint inhibitors derive efficacy from this cancer-immune interface; however, immune-related adverse events from systemic blockade remain a major challenge. To address this need for potent, tumor-specific immunotherapies, we developed Tumor-Immune Cell TArgeting Chimeras (TICTACs) that selectively deplete immune checkpoint receptors such as SIRP from TAM surfaces. These chimeras consist of a synthetic ligand targeting CD206, a TAM marker, conjugated to a non-blocking antibody that binds without inhibiting the checkpoint receptor. By engaging CD206, which constitutively recycles between the plasma membrane and early endosomes, TICTACs drive robust checkpoint degradation in CD206high macrophages, with no effect on CD206low cells. This decoupling of antibody selectivity from blocking function presents a new paradigm for tumor-specific immunotherapies.

biochemistry↗

Three-dimensional markerless motion capture of multiple freely behaving monkeys for automated characterization of social behavior

Given their high sociality and close evolutionary distance to humans, monkeys are an essential animal model for unraveling the biological mechanisms underlying human social behavior and elucidating the pathogenesis of diseases exhibiting abnormal social behavior. However, behavioral analysis of naturally behaving monkeys requires manual counting of various behaviors, which has been a bottleneck due to problems in throughput and objectivity. Here, we developed a three-dimensional markerless motion capture system that utilized multi-view data for robust tracking of individual monkeys and accurate reconstruction of the three-dimensional poses of multiple monkeys living in groups. Validation analysis in two monkey groups revealed that the system enabled the characterization of individual social dispositions and relationships through automated detection of various social events. Analyses of social looking facilitated the investigation of adaptive behaviors in a social group. These results suggest that this motion capture system will significantly enhance our ability to analyze primate social behavior.

animal behavior and cognition↗

The SOCE system is critical for membrane bleb formation to drive avian primordial germ cell migration

Amoeboid cell migration is driven by the specialized cell protrusion, membrane bleb. A recent in vitro analysis of bleb formation using a cancer cell line showed that store-operated calcium entry (SOCE)-mediated local elevation of Ca2+ concentration triggers bleb formation, but it remains unknown how commonly this system is utilized in the bleb formation and bleb-driven cell migration in vivo. We demonstrate with avian primordial germ cell (PGC) model that that chick PGCs use SOCE, triggered by stem cell factor (SCF), to induce bleb formation and that it is essential for in vivo migration including trans-endothelial and mesenchymal cell migration. Our discovery also provides insight into the correlation between cancer metastasis/invasion and SOCE-mediated bleb formation.

developmental biology↗

CUX2 mutations in temporal lobe epilepsy; increased kainate susceptibility and excitatory input to hippocampus in deficient mice

CUX2 gene encodes a transcription factor that controls neuronal proliferation, dendrite branching and synapse formation, locating at the epilepsy-associated chromosomal region 12q24 that we previously identified by a genome-wide association study (GWAS) in Japanese population. A CUX2 recurrent de novo variant p.E590K has been described in patients with rare epileptic encephalopathies and the gene is a candidate for the locus, however the mutation may not be enough to generate the genome-wide significance in the GWAS and whether CUX2 variants appear in other types of epilepsies and physiopathological mechanisms are remained to be investigated. Here in this study, we conducted targeted sequencings of CUX2, a paralog CUX1 and its short isoform CASP harboring a unique C-terminus on 271 Japanese patients with a variety of epilepsies, and found that multiple CUX2 missense variants, other than the p.E590K, and some CASP variants including a deletion, predominantly appeared in patients with temporal lobe epilepsy (TLE). The CUX2 variants showed abnormal localization in human cell culture analysis. While wild-type CUX2 enhances dendritic arborization in fly neurons, the effect was compromised by some of the variants. Cux2- and Casp-specific knockout mice both showed high susceptibility to kainate, increased excitatory cell number in the entorhinal cortex, and significant enhancement in glutamatergic synaptic transmission to the hippocampus. CASP and CUX2 proteins physiologically bound to each other and co-expressed in excitatory neurons in brain regions including the entorhinal cortex. These results suggest that CUX2 and CASP variants contribute to the TLE pathology through a facilitation of excitatory synaptic transmission from entorhinal cortex to hippocampus.

neuroscience↗

Airway tissue stem cells reutilize the embryonic proliferation regulator, Tgfβ-Id2 axis, for tissue regeneration

During development, quiescent basal stem cells are derived from proliferative primordial progenitors through the cell cycle slowdown. In contrast, quiescent basal cells contribute to tissue repair during adult tissue regeneration by shifting from slow-cycling to proliferating and subsequently back to slow-cycling. Although sustained basal cell proliferation results in tumorigenesis, the molecular mechanisms regulating these transitions remain unknown. Using temporal single-cell transcriptomics of developing murine airway progenitors and in vivo genetic validation experiments, we found that Tgf{beta} signaling slowed down cell cycle by inhibiting Id2 expression in airway progenitors and contributed to the specification of slow-cycling basal cell population during development. In adult tissue regeneration, reduced Tgf{beta} signaling restored Id2 expression and initiated epithelial regeneration. Id2 overexpression and Tgfbr2 knockout enhanced epithelial proliferation; however, persistent Id2 expression in basal cells drove hyperplasia at a rate that resembled a precancerous state. Together, the Tgf{beta}-Id2 axis commonly regulates the proliferation transitions in airway basal cells during development and regeneration, and its fine-tuning is critical for normal regeneration while avoiding basal cell hyperplasia.

developmental biology↗

Single cell transcriptomics reveals a signaling roadmap coordinating endoderm and mesoderm lineage diversification during foregut organogenesis

Visceral organs, such as the lungs, stomach, liver and pancreas, are derived from the fetal foregut through a series of inductive interactions between the definitive endoderm (DE) and the surrounding splanchnic mesoderm (SM). While patterning of DE lineages has been fairly well studied, paracrine signaling controlling SM regionalization and how this is coordinated with the epithelial identity during organogenesis is obscure. Here we used single cell transcriptomics to generate a high-resolution cell state map of the embryonic mouse foregut. This uncovered an unexpected diversity in the SM cells that developed in close register with the organ-specific epithelium. From these data, we inferred a spatiotemporal signaling roadmap of the combinatorial endoderm-mesoderm interactions that orchestrate foregut organogenesis. We validated key predictions with mouse genetics, showing the importance of endoderm-derived signals in mesoderm patterning. Finally, leveraging the signaling road map we generated different SM subtypes from human pluripotent stem cells (hPSCs), which previously have been elusive. The single cell data can be explored at: https://research.cchmc.org/ZornLab-singlecell.

developmental biology↗

Induction of tracheal mesoderm and chondrocyte from pluripotent stem cells in mouse and human.

The periodic cartilage and smooth muscle structures in mammalian trachea are derived from tracheal mesoderm, and tracheal malformations result in serious respiratory defects in neonates. Here we show that canonical Wnt signaling in mesoderm is critical to confer trachea mesenchymal identity in human and mouse. Loss of {beta}-catenin in fetal mouse mesoderm caused loss of Tbx4+ tracheal mesoderm and tracheal cartilage agenesis. The Tbx4 expression relied on endodermal Wnt activity and its downstream Wnt ligand but independent of known Nkx2.1-mediated respiratory development, suggesting that bidirectional Wnt signaling between endoderm and mesoderm promotes trachea development. Repopulating in vivo model, activating Wnt, Bmp signaling in mouse embryonic stem cell (ESC)-derived lateral plate mesoderm (LPM) generated tracheal mesoderm containing chondrocytes and smooth muscle cells. For human ESC-derived LPM, SHH activation was required along with Wnt to generate proper tracheal mesoderm. Together, these findings may contribute to developing applications for human tracheal tissue repair.

developmental biology↗

Genome-wide association meta-analysis identifies novel GP2 gene risk variants for pancreatic cancer in the Japanese population

The etiology of pancreatic cancer remains largely unknown. Here, we report the results of a meta-analysis of three genome-wide association studies (GWASs) comprising 2,039 pancreatic cancer cases and 32,592 controls, the largest sample size in the Japanese population. We identified 3 (13q12.2, 13q22.1, and 16p12.3) genome-wide significant loci (P<5.0x10-8) and 4 suggestive loci (P<1.0x10-6) for pancreatic cancer. Of these risk loci, 16p12.3 is novel; the lead SNP maps to rs78193826 (odds ratio (OR)=1.46, 95% CI=1.29-1.66, P=4.28x10-9), an Asian-specific, nonsynonymous glycoprotein 2 (GP2) gene variant predicted to be highly deleterious. Additionally, the gene-based GWAS identified a novel gene, KRT8, which is linked to exocrine pancreatic and liver diseases. The identified GP2 gene variants were pleiotropic for multiple traits, including type 2 diabetes, hemoglobin A1c (HbA1c) levels, and pancreatic cancer. Mendelian randomization analyses corroborated causality between HbA1c and pancreatic cancer. These findings suggest that GP2 gene variants are associated with pancreatic cancer susceptibility in the Japanese population, prompting further functional characterization of this locus.

genetics↗

Cerebral amyloid angiopathy and comparative analysis of amyloid-β protein in birds

Senile plaques and cerebral amyloid angiopathy (CAA) are well-documented in various mammals, and several species even exhibit neurofibrillary tangle (NFT). However, we know far less about whether such symptoms are present in birds. Therefore, here we clarified the occurrence and pathogenesis of avian a{beta}-related lesions, analyzing the a{beta} amino-acid sequence across 28 birds at multiple life stages, representing 15 species, 14 genera, and 9 nine families. We also determined the expected a{beta} amino-acid sequence after comparing data from the brains of nine birds (seven species) with publicly available NCBI data. We observed CAA and senile plaque-like deposition only in a female Amazon parrot, estimated to be around 30-40 years old. We identified two A{beta} depositions (40 and 42) in the same location that correspond to A{beta} 6-42. Additionally, we observed severe A{beta} deposition, accompanied by severe hemorrhaging, in blood vessels of the superficial and deep portions of the brain. These lesions were directly related to the cause of death. Of 40 bird species, 36 exhibited type 1 A{beta} amino-acid sequences, similar to humans. Given that all of these birds were old, our results suggest that A{beta} is deposited primarily as CAA as the animals age. This report is the first clinically based description of CAA in birds. Interspecific variation likely exists because we identified species that did not exhibit A{beta} deposition even when the birds are old enough. However, even birds of the same taxonomic status differed in whether they possessed or lacked A{beta} deposition. Thus, other factors besides A{beta} amino-acid sequence could influence this symptom.

pathology↗