Issues
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Cover image
Cover Image
Cover: Craniofacial skeleton preparation after Alcian Blue staining of a 4-day-old Xenopus tadpole. Craniofacial structures are mainly derived from migratory neural crest cells. The migration of neural crest cells is dependent on dermatan sulfate. A failure in proper neural crest cell migration might explain the craniofacial anomalies and other congenital malformations in musculocontractural Danlos–Ehlos syndrome, which results from a defect in dermatan sulfate biosynthesis. See article by Gouignard et al. on page 607. ‘Dermatan sulfate in neural crest cell migration’ by N. Gouignard is licenced under a Creative Commons Attribution 4.0 International licence.
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EDITORIAL
Modeling human diseases: an education in interactions and interdisciplinary approaches
Summary: Animal models have greatly facilitated the process of drug discovery. Len Zon draws on his own experiences to highlight how a cross-model, interdisciplinary approach can bring basic findings to the clinic.
SPECIAL ARTICLE
Show and tell: disclosure and data sharing in experimental pathology
Summary: Reproducibility of findings in experiments using model organisms has recently become a source of concern, particularly for translational science. We discuss factors affecting the interpretation and reliability of experimental pathology findings in the mouse, and how disclosure and transparent reporting are crucial for replicability.
RESEARCH ARTICLES
Musculocontractural Ehlers–Danlos syndrome and neurocristopathies: dermatan sulfate is required for Xenopus neural crest cells to migrate and adhere to fibronectin
Editors' choice: In the Xenopus neural crest, dermatan sulfate is essential for cell migration in vivo and cell adhesion to fibronectin, which might have implications for musculocontractural Ehlers–Danlos syndrome and cancer.
Identification of benzopyrone as a common structural feature in compounds with anti-inflammatory activity in a zebrafish phenotypic screen
Summary: Zebrafish inflammation screen identifies a new series of structurally related compounds with combined anti-inflammatory and pro-resolution activity, and reveals a previously unknown mechanism of action of clinical cromones.
Histone lysine crotonylation during acute kidney injury in mice
Summary: We have assessed the effect of the epigenetic post-translational modification histone crotonylation during kidney injury in vivo and in cell culture, and the involvement of PGC-1α and SIRT3 in the process.
Two different pathogenic mechanisms, dying-back axonal neuropathy and pancreatic senescence, are present in the YG8R mouse model of Friedreich’s ataxia
Summary: Frataxin deficiency induces different pathogenic mechanisms in the nervous system and pancreas in a YG8R mouse model of Friedreich's ataxia (FRDA). Thus, the degenerative process in FRDA is determined by the cell type.
A novel Drosophila model of TDP-43 proteinopathies: N-terminal sequences combined with the Q/N domain induce protein functional loss and locomotion defects
Summary: An engineered TDP-43 construct can be used to induce TDP-43 aggregation in Drosophila, providing a model that could be useful for characterization of pathogenetic mechanisms and drug screening.
Cellular dynamics of regeneration reveals role of two distinct Pax7 stem cell populations in larval zebrafish muscle repair
Summary: Tracking muscle precursor cells during wound repair with confocal time-lapse microscopy reveals that two myoblast populations fuse and contribute differentially to regeneration, one initiating and the other growing fibres.
High-throughput screening for modulators of ACVR1 transcription: discovery of potential therapeutics for fibrodysplasia ossificans progressiva
Summary: We describe the identification of dipyridamole as a potential therapeutic tool for FOP, through a series of in vitro and in vivo assays to screen and validate FDA-approved compounds.
Muscles provide protection during microbial infection by activating innate immune response pathways in Drosophila and zebrafish
Summary: Using fruit fly and zebrafish models, we show that skeletal muscles are immune responsive tissues; they mount innate immune responses during bacterial infection – an evolutionarily conserved defense mechanism.
TLR-mediated albuminuria needs TNFα-mediated cooperativity between TLRs present in hematopoietic tissues and CD80 present on non-hematopoietic tissues in mice
Summary: Systemic TNFα mediates myeloid cell and podocyte cross-talk to cause LPS-induced mouse microalbuminuria, a partial model of human nephrotic syndrome, pointing to potential adjunct therapeutic approaches.
DMM Journal Meeting 2024: Pre-clinical Modelling of Human Genetic Disease and Therapy

Register your interest for our 2024 Journal Meeting. Rapid advances in gene editing and genetic technologies have revolutionised our ability to model human genetic disease and provided new hope for gene therapies. At this Meeting, we will present the very latest advances in modelling human genetic disease. Find more information here.
A new call for papers is underway

Showcase your latest research in our upcoming special issue Translating Multiscale Research in Rare Disease, coordinated by DMM Editors Monica Justice, Karen Liu and Monkol Lek, and Guest Editor Kate Rauen. The deadline for submitting articles to the special issue is Monday 6 November 2023.
Moving towards heart success – Disease Models & Mechanisms Special Issue

DMM's most recent special issue compiles articles that aim to move heart failure to heart success by fundamentally addressing the roots of failure to identify curative strategies.
Adult zebrafish as advanced models of human disease

Editor-in-Chief Liz Patton and Editorial Board member Rich White discuss the unique advantages of adult zebrafish for studying human disease biology. Read the Open Access Editorial here.
The Forest of Biologists

The Forest of Biologists is a biodiversity initiative created by The Company of Biologists, with support from the Woodland Trust. For every Research and Review article published in Disease Models & Mechanisms a native tree is planted in a UK forest. In addition to this we are protecting and restoring ancient woodland and are dedicating these trees to our peer reviewers. Visit our virtual forest to learn more.