DaniocellDesktop: for Interactive Reanalysis of Wild-Type Zebrafish Single-Cell Genomic Data.

DaniocellDesktop is a cross-platform interactive desktop application designed to facilitate reanalysis of a previously published 462,243-cell single-cell RNAseq dataset that profiled cell-type-specific gene expression across the first 5 days of wild-type zebrafish development. DaniocellDesktop enables custom redefinition of cell populations, identification of differentially expressed genes, and generation of several types of publication-ready plots to show gene expression patterns, gene co-expression patterns, and gene expression over time within these previously published data without requiring any specific programming knowledge. This software is available from https://daniocell.

Liver-specific loss of perturbs lipid metabolism and hepatocyte integrity.

The autophagy-related protein ATG9A is integral to cellular autophagy and lipid mobilization, yet its importance in mammalian physiology remains underexplored. Using a liver-specific conditional knockout (-cKO) mouse model, we uncovered critical insights into the physiological function of ATG9A in this organ. -cKO mice exhibited hepatomegaly, abnormal hepatocyte morphology, mitochondrial fragmentation, and lipid droplet accumulation.

Growth trajectories and risk of overweight and obesity in twins and singletons in the UK and USA: analysis of prospective national cohort data.

Background: Twin and singleton growth might be adaptively different to maximise survival in a shared intrauterine environment, translating to smaller average twin birthweight. We aimed to compare growth trajectories and risks of overweight and obesity between twins and singletons.

Methods: We leveraged height, weight, and BMI data from two large and ongoing nationally representative cohorts, the Millennium Cohort Study (MCS) and the National Longitudinal Survey of Youth-Child and Young Adult (NLSY-CYA), that have prospectively collected data from children born in the UK between Sept 1, 2000, and Jan 11, 2002, and participants born in the USA between December, 1970, and January, 2008, respectively, every 2-3 years from birth to adulthood.

Multiple child caregivers and multiple cognitive caregiving practices: Associations with early childhood development in 51 low- and middle-income countries.

Five issues about three cognitive caregiving practices are addressed in mothers, fathers, and children's other caregivers in nationally representative samples from 51 low- and middle-income countries with 159,959 36- to 59-month-old children. The five issues include base rates of cognitive caregiving practices of mothers, fathers, and other caregivers of young children, associations of cognitive caregiving practices of the three caregivers with children's development, associations among the three caregivers' cognitive caregiving practices, comparison of the three caregivers' cognitive caregiving practices with girls and boys, and assessments of how overall national development relates to the three caregivers' cognitive caregiving practices. The data addressing these five issues derive from the maternal reports in UNICEF's Multiple Indicator Cluster Surveys.

The Allele is a Missense Mutation Within the Predicted Tryptophan 2,3-dioxygenase Protein Domain of .

Many loss-of-function mutations in the ( ) gene have been described. However, the causal mutation in the common allele is unknown. We sequenced different alleles ( , , and ) to identify candidate mutations.

A novel and evolutionarily conserved inhibitory circuit selectively regulates dentate gyrus mossy cell function.

The mammalian dentate gyrus contributes to mnemonic function by parsing similar events and places. The disparate activity patterns of mossy cells and granule cells are believed to enable this function yet the mechanisms that drive this circuit dynamic remain elusive. We identified a novel inhibitory interneuron subtype, characterized by VGluT3 expression, with overwhelming target selectivity for mossy cells while also revealing that CCK, PV, SST and VIP interneurons preferentially innervate granule cells.

BLOC-1 and BORC: Complex regulators of endolysosomal dynamics.

Endolysosomes are dynamic organelles that undergo movement along the cytoskeleton, fusion, fission, and tubulation during their lifetime. These processes are regulated by complex molecular machineries, including the structurally related hetero-octameric complexes BLOC-1 and BORC. BLOC-1 associates with early endosomes to mediate the biogenesis of lysosome-related organelles (LROs), such as melanosomes and platelet dense bodies.

Ambient air pollution during developmental windows of spermatogenesis and semen quality among an infertility treatment seeking population.

Prior studies have observed impacts of air pollution on semen quality, but timing of exposure during developmental windows of spermatogenesis and impacts of low-to-moderate air pollution is less well understood. We examined the relation between air pollution and semen quality in the Folic Acid and Zinc Supplementation Trial (2013-2018), which enrolled male partners of couples seeking infertility treatment in the Salt Lake City, Utah region (n = 2015). Semen quality parameters were assessed at baseline, 2-, 4-, and 6-months follow-up.

Zika in Infants and Pregnancy (ZIP) study: results from a prospective international cohort study of prenatal Zika virus infection and adverse fetal and infant outcomes.

Background: Before Zika virus (ZIKV) infections were observed in the Americas, an association between ZIKV and microcephaly or other congenital malformations was not well documented. Initial reports suggested strong associations between ZIKV and congenital malformations, but plausible estimates of causal effects from prospective studies with adequate sample size and covariate data were few.

Methods: From 2016-2018, the Zika in Infants and Pregnancy (ZIP) study enrolled pregnant people before 18 weeks gestation or with confirmed symptomatic ZIKV in a prospective cohort across 10 sites in South and Central America, and in Puerto Rico.

Prosocial Behavior and School Performance in the Transition to Adolescence: A Multicultural Study.

The present study explored the bidirectional longitudinal associations between prosocial behavior and school performance during adolescence in six countries (Colombia, Italy, Jordan, the Philippines, Thailand, and the United States). A total sample of 884 adolescents (T1: = 10.34 years, = 0.

Bayesian additive tree ensembles for composite quantile regressions.

In this paper, we introduce a novel approach that integrates Bayesian additive regression trees (BART) with the composite quantile regression (CQR) framework, creating a robust method for modeling complex relationships between predictors and outcomes under various error distributions. Unlike traditional quantile regression, which focuses on specific quantile levels, our proposed method, composite quantile BART, offers greater flexibility in capturing the entire conditional distribution of the response variable. By leveraging the strengths of BART and CQR, the proposed method provides enhanced predictive performance, especially in the presence of heavy-tailed errors and non-linear covariate effects.

Expansion of the Phenotype of Lymphatic Anomalies Caused by Somatic Activating BRAF Variant.

Background: The somatic activating variant in BRAF (p.V600E) was recently described as a novel cause of macrocystic head and neck lymphatic malformations in three individuals. Other recent studies profiling the genetic causes of more complex lymphatic anomalies identified this same pathogenic BRAF variant.

Maternal Adherence to Healthy Dietary Patterns During Pregnancy and Gestational Weight Gain.

: Suboptimal gestational weight gain (GWG) has been linked to increased risks of adverse maternal outcomes. Evidence linking diet in pregnancy to GWG remains limited. We assessed relationships between adherence to five dietary patterns (Planetary Health Diet [PHD], Dietary Approaches to Stop Hypertension [DASH], Alternate Mediterranean Diet [AMED], Healthy Eating Index [HEI], and Alternate Healthy Eating Index [AHEI]) and 2009 Institute of Medicine GWG categories.

Development of the Visual Analysis of Form and Contour.

Background/objectives: A common approach to investigating visual form processing is through studying responses to visual stimuli that comprise illusory contours. Such stimuli induce contours where none exist physically and thus reveal the constructive nature of visual perception and the conditions that engender it. The present work used IC stimuli to study the development of visual form detection and extraction in infants and adults.

The Effect of Experimentally Induced Cognitive Fatigue on Energy Intake Among Youth With and Without Recent Reported Dietary Restraint.

Objective: Resource-based models of self-regulation posit that cognitive fatigue, or the depletion of cognitive resources, may be an impetus for self-regulatory difficulties, including overeating. Studies of adults indicate that cognitive fatigue may increase energy intake only among adults who report engaging in dietary restraint (DR). The current study examined if DR similarly moderates the effects of cognitive fatigue on energy intake in youth.

Validity and reliability of the Mini International Neuropsychiatric Interview in Sub-Saharan Africa: a cross-country comparison study.

Background: Diagnostic tools, such as the Mini International Neuropsychiatric Interview (MINI) 7.0.2 and the Structured Clinical Interview for the DSM-5 (SCID), aim to increase the validity and reliability of diagnostic assessment.

Disentangling consequences of self-perceived weight from excess adiposity in metabolically healthy overweight/obesity.

Background: Greater self-perceived weight is detrimental to cardiometabolic health among people with overweight/obesity. However, it is impractical to test and unknown whether weight-related psychosocial factors, like self-perceived weight, independently affect cardiometabolic health apart from physiological dysregulation produced by excess adiposity among people with overweight/obesity. Metabolically healthy overweight/obesity (MHOv/Ob) involves overweight/obesity, but with the absence of metabolic dysfunction.

LC3 and GABARAP independent autophagy of misfolded procollagen in mouse osteoblasts.

Bone synthesis should depend on autophagy because over 10% of type I procollagen (PC1) - a heterotrimer of COL1A1 and COL1A2 chains and the precursor of the main bone matrix molecule - is misfolded and rerouted from osteoblast endoplasmic reticulum (ER) to lysosomes. However, osteoblast-specific macroautophagy knockouts in mice have produced only mild bone effects. To reconcile these observations, we compared how hypomorphic expression and a conditional knockout (cKO) of - encoding a protein required for autophagosome formation - affected versus wild-type osteoblasts and .

"To Err is Human: Lessons from Prolapse Studies to Identify and Strategies to Reduce Data Errors".

Objective: The aim of this manuscript is to describe the quality improvement auditing process developed by the Pelvic Floor Disorders Network of the Eunice Kennedy Shriver NICHD for assessing the primary outcome of pelvic organ prolapse quantification (POP-Q) measurements in their prolapse research trials and provide the reader with the most iterative POP-Q form.

Methods: We reviewed the Pelvic Floor Disorders Network steering committee minutes, data coordinating center audit reports, and the case report forms for studies related to prolapse and summarized findings.

Results: Errors found were related to data entry and documentation of pelvic organ prolapse quantification data with sign errors and inaccurate Ba/Bp point assessments being most common.

Combination nitazoxanide and auranofin treatment has synergistic anticancer activity in anaplastic thyroid cancer through enhanced activation of oxidative stress that leads to apoptosis.

Anaplastic thyroid cancer (ATC) has one of the highest mortality rates of all human malignancies and has no cure. We used combination drug matrix screening of highly active compounds and identified that combination of nitazoxanide and auranofin was one of those with the highest synergistic anticancer activity. We investigated its synergistic anticancer activity and mechanism of action in preclinical ATC models.

The hereditary spastic paraplegia type 21 (SPG21) protein is a RAB7A effector that promotes mTORC1-catalyzed TFEB phosphorylation and cytoplasmic retention.

Hereditary spastic paraplegia type 21 (SPG21) is an inherited neurological disorder caused by biallelic mutations in the gene, which encodes a protein named SPG21 or maspardin. Herein, we report that the SPG21 protein localizes to endolysosomes through interaction with the GTP-bound form of RAB7A. Disease-associated variants reduce expression of SPG21 and disrupt its endolysosomal localization in both non-neuronal cells and neurons.

Phase II Study of Lu-DOTATATE for Progressive Metastatic Pheochromocytomas and Paragangliomas: Interim Analysis of Efficacy, Safety, and Biomarkers.

Purpose: Lu-DOTA(0)-Tyr(3)-octreotate (Lu-DOTATATE) is a somatostatin receptor (SSTR)-targeting radiopharmaceutical that shows promise for treating metastatic pheochromocytomas/paragangliomas (PPGLs), a rare SSTR-expressing tumor.

Methods: In the first stage of this two-stage Simon phase II trial, 36 PPGL patients with RECIST 1.1 progression within 12 months were prospectively recruited into two genetic cohorts (succinate dehydrogenase []-mutated apparent sporadic, 18 per cohort) and treated with four cycles of Lu-DOTATATE.

Programmed meiotic errors facilitate dichotomous sperm production in the silkworm, .

The goal of meiosis is typically to produce haploid gametes (eggs or sperm). Failure to do so is catastrophic for fertility and offspring health. However, Lepidopteran (moths and butterflies) males produce two morphs of sperm: nucleated (eupyrene) sperm which fertilize the egg, and anucleated (apyrene) sperm, both of which are essential for fertilization.

prdm1a drives a fate switch between hair cells of different mechanosensory organs.

Vertebrate inner ear mechanosensory hair cells detect sound and gravitational forces. Additionally, fishes have homologous lateral line hair cells in the skin that detect water vibrations for orientation and predator avoidance. Hair cells in the lateral line and ear of fishes and other non-mammalian vertebrates regenerate readily after damage, but mammalians lack this ability, causing deafness and vestibular defects.