Issues
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Cover image
Cover Image
Cover: The rate of protein synthesis in live Drosophila wings is measured by using a fluorescent puromycin analogue incorporated into nascent proteins (red). Too much or too little of the nucleoporin Nup98 can lead to defects in protein synthesis, thereby contributing to signalling changes in tissues that exhibit Nup98 mis-regulation. GFP expression in the posterior developing wing is shown in green, nuclei are stained in blue. See article by Pulianmackal et al. (dmm049234). Cover image is licensed under a Creative Commons Attribution 4.0 International license.
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EDITOR'S CHOICE
EDITORIAL
The power of Drosophila in modeling human disease mechanisms
Summary: This Editorial encourages us to embrace the power of the fly in studying human disease and highlights how Drosophila studies can be integrated with research in other species to further our understanding of human biology.
A MODEL FOR LIFE
PERSPECTIVE
Embracing complexity in Drosophila cancer models
Summary: Comorbidities and tumour genetic diversity more accurately reflect the cancer patient landscape but are largely neglected in animal models. Drosophila holds the potential to address these complexities to better understand their impacts on cancer development.
REVIEWS
Investigating local and systemic intestinal signalling in health and disease with Drosophila
Summary: We outline work in the fruit fly Drosophila melanogaster that has contributed knowledge on local and whole-body signalling coordinated by the adult intestine, and discuss its implications in intestinal pathophysiology and associated systemic dysfunction.
Cancer cachexia: lessons from Drosophila
Summary: Drosophila tumor models have contributed to our knowledge of cancer cachexia. Here, we review current knowledge on Drosophila cachectic factors, their effects on host organs and metabolism, and discuss open questions for future studies.
RESEARCH ARTICLES
Insights into cisplatin-induced neurotoxicity and mitochondrial dysfunction in Caenorhabditis elegans
Summary: Using Caenorhabditis elegans to explore neuronal and metabolic factors influencing cisplatin toxicity revealed that dopamine treatment protects against the dose-dependent neurotoxicity caused by cisplatin.
The EDA-deficient mouse has Zymbal's gland hypoplasia and acute otitis externa
Summary: Ectodysplasin-deficient mice have growth retardation of the auditory sebaceous Zymbal's gland and are predisposed to spontaneous bacterial infection of the outer ear canal by opportunistic pathogens.
Aberrant RET expression affects normal mammary gland post-lactation transition, enhancing cancer potential
Summary: We show that RET activation stimulates Stat3 signaling in mammary epithelial cell culture and in vivo during post-lactation transition, demonstrating that the RET receptor participates in the post-lactation transition priming tumorigenesis.
Zebrafish drug screening identifies candidate therapies for neuroprotection after spontaneous intracerebral haemorrhage
Summary: We used a zebrafish larval model to perform drug screening to identify novel and translational compounds for ameliorating brain damage after a haemorrhage, which may support neuroprotective therapeutic development.
Dysregulated mitochondrial metabolism upon cigarette smoke exposure in various human bronchial epithelial cell models
Summary: Exposure of primary human bronchial epithelial cells to smoke affected mitochondria differently in various exposure and culture models, highlighting the importance of tailoring the model to the research question.
Latent TGFβ-binding proteins 1 and 3 protect the larval zebrafish outflow tract from aneurysmal dilatation
Summary: This study describes one of the first zebrafish models of thoracic aortic aneurysm, arising from mutations in ltbp1 and ltbp3, genes that regulate TGFβ signaling.
Exercise suppresses mouse systemic AApoAII amyloidosis through enhancement of the p38 MAPK signaling pathway
Summary: Interval exercise training delays the progression of mouse systemic amyloidosis, with potential exercise-induced mechanisms that activate the anti-amyloid chaperone HSPB1 enhanced by the p38 MAPK signaling pathway.
Misregulation of Nucleoporins 98 and 96 leads to defects in protein synthesis that promote hallmarks of tumorigenesis
Editor's choice: Compromising Nucleoporins 98 and 96 leads to defects in protein synthesis and stress signaling via JNK that triggers compensatory and apoptosis-induced proliferation.
A protein-trap allele reveals roles for Drosophila ATF4 in photoreceptor degeneration, oogenesis and wing development
Summary: A protein-trap line for Drosophila ATF4, encoded by cryptocephal, reveals how this integrated stress response transcription factor responds to physiological stress, and demonstrates its role in Drosophila development and retinal degeneration.
Parietal epithelial cells maintain the epithelial cell continuum forming Bowman's space in focal segmental glomerulosclerosis
Summary: Histological analysis of rat and human kidneys reveals a novel role for parietal epithelial cells (PECs) in glomerulosclerosis. PECs seem to restore the glomerular epithelial continuum, which may avert further loss of glomerular function.
The schizophrenia risk gene Map2k7 regulates responding in a novel contingency-shifting rodent touchscreen gambling task
Summary: This work identifies in mice a single, specific gene variation linked to schizophrenia risk (hemizygosity for genetic deletion of MKK7) that produces a specific cognitive impairment exhibited by patients with schizophrenia.
A Matrigel-based 3D construct of SH-SY5Y cells models the α-synuclein pathologies of Parkinson's disease
Summary: This study provides a convenient 3D model of Parkinson's disease (PD), which recapitulates α-synuclein pathologies in human cells and could be used to investigate PD mechanisms and screen drugs.
Novel preclinical model for CDKL5 deficiency disorder
Summary: Characterisation of a novel loss-of-function zebrafish model for CDKL5 deficiency disorder, containing a nonsense mutation, demonstrates its utility for investigating disease aetiology and allowing high-throughput screening for potentially effective therapies.
Post-synaptic scaffold protein TANC2 in psychiatric and somatic disease risk
Summary: Disruption of mouse Tanc2 causes brain and liver abnormality, increasing psychiatric and somatic disease risk long term, highlighting the benefit of holistic diagnosis and treatment approaches for human neurodevelopmental disorder.
Validation of DE50-MD dogs as a model for the brain phenotype of Duchenne muscular dystrophy
Summary: The DE50-MD canine model of Duchenne muscular dystrophy (DMD) shows cognitive and brain biochemical changes secondary to dystrophin deficiency in the brain, and is a relevant pre-clinical model to develop therapies for DMD.
A novel and efficient murine model of Bietti crystalline dystrophy
Summary: A novel and more effective Cyp4v3 knockout preclinical mouse model for Bietti crystalline dystrophy, which will serve as a useful tool for investigating the pathogensis of the disease in vivo.
Development and characterization of cell models harbouring mtDNA deletions for in vitro study of Pearson syndrome
Summary: Comparison of three different cell models from Pearson syndrome patients harbouring mitochondrial DNA deletions, which showed a severe failure in OXPHOS function, thus confirming their usefulness for therapeutic research.
Abnormal brain development of monoamine oxidase mutant zebrafish and impaired social interaction of heterozygous fish
Summary: We assessed developmental, neurochemical and behavioral alterations displayed by mao+/− and mao−/− zebrafish, establishing that these model organisms are promising tools to study the consequences of MAOA/B deficiency.
Treatment of age-related visual impairment with a peptide acting on mitochondria
Summary: Age-related decline in vision in mice is substantially prevented or restored in response to treatment with a peptide that comprises mitochondrial affinity and improves mitochondrial function.
RESOURCE ARTICLES
Molecular Subtyping Resource: a user-friendly tool for rapid biological discovery from transcriptional data
Summary: We developed an open source and user-friendly data analysis tool, Molecular Subtyping Resource (MouSR), which enables users to perform rapid transcriptomic analyses and comprehensive interrogation of the signalling underpinning transcriptional datasets, potentially facilitating biological discovery.
Longitudinal multimodal imaging-compatible mouse model of triazole-sensitive and -resistant invasive pulmonary aspergillosis
Summary: A novel reproducible longitudinal multimodal imaging-compatible neutropenic mouse model of invasive pulmonary aspergillosis provides increased early fungal detection through novel red-shifted luciferase-expressing triazole-susceptible and -resistant Aspergillus fumigatus strains, and boosted bioluminescence.
FIRST PERSON
CORRECTION
New Special Issue: Translating Multiscale Research in Rare Disease. Edited by Monica Justice, Monkol Lek, Karen Liu and Kate Rauen.
This special issue features original Research, Resources & Methods and Review-type articles that aim to interrogate the mechanisms of rare diseases to foster meaningful clinical progress in their diagnosis and treatment.
The role of the International Society for Stem Cell Research (ISSCR) guidelines in disease modelling
The ISSCR provides comprehensive guidelines and standards for using human stem cells in biomedical research. In this Editorial, Cody Juguilon and Joseph Wu discuss how and why these should be incorporated in disease modelling research.
Subject collection: Building advocacy into research
DMM’s series - Building advocacy into research - features interviews, ‘The Patient’s Voice’, with patients and advocates for a range of disease types, with the aim of supporting the highest quality research for the benefit of all patients affected by disease.
Read & Publish Open Access publishing: what authors say
We have had great feedback from authors who have benefitted from our Read & Publish agreement with their institution and have been able to publish Open Access with us without paying an APC. Read what they had to say.
Biologists @ 100 - join us in Liverpool in March 2025
We are excited to invite you to a unique scientific conference, celebrating the 100-year anniversary of The Company of Biologists, and bringing together our different communities. The conference will incorporate the Spring Meetings of the BSCB and the BSDB, the JEB Symposium Sensory Perception in a Changing World and a DMM programme on antimicrobial resistance. Find out more and register your interest to join us in March 2025 in Liverpool, UK.