Amanda Teixeira, Cassandra Millet-Boureima, Joshua A Moore, Loydie A Jerome-Majewska
{"title":"Secretopathies emerge as a new class of neurocristopathies.","authors":"Amanda Teixeira, Cassandra Millet-Boureima, Joshua A Moore, Loydie A Jerome-Majewska","doi":"10.1002/dvdy.70140","DOIUrl":"https://doi.org/10.1002/dvdy.70140","url":null,"abstract":"<p><p>Neural crest cells are a transient embryonic population of cells that give rise to a wide range of structures, including craniofacial cartilage and bone, peripheral neurons and glia, as well as components of the cardiac outflow tract, among others. Proper formation, migration and differentiation of NCCs require tightly regulated secretion of signaling molecules, extracellular matrix components and membrane proteins, rendering NCCs particularly reliant on a highly efficient and precisely regulated secretory pathway. Disruption of this pathway has emerged as an important, yet underappreciated, mechanism underlying developmental disease. Mutations in genes encoding components of the secretory machinery have been linked to congenital disorders that affect neural crest-derived tissues, suggesting that impaired intracellular trafficking and cargo export can compromise NCC function. In this review, we propose that such disorders represent a distinct subclass of neurocristopathies, termed secretopathies, in which defects in secretory pathway drive neural crest-associated phenotypes. We further highlight TMED proteins as key cargo receptors within the early secretory pathway whose dysfunction may impact NCC secretory output. Recognizing secretopathies as a novel category of neurocristopathies provides a new framework for understanding the molecular basis of neural crest-related developmental disorders.</p>","PeriodicalId":11247,"journal":{"name":"Developmental Dynamics","volume":" ","pages":""},"PeriodicalIF":1.5,"publicationDate":"2026-05-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147812270","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Differential sensitivity of midline development to mitosis during and after primitive streak extension.","authors":"Zhiling Zhao, Rieko Asai, Takashi Mikawa","doi":"10.1002/dvdy.70045","DOIUrl":"10.1002/dvdy.70045","url":null,"abstract":"<p><strong>Background: </strong>Midline establishment is a fundamental process during early embryogenesis for Bilaterians. Midline morphogenesis in non-amniotes can occur without mitosis, through Planar Cell Polarity (PCP) signaling. By contrast, amniotes utilize both cellular processes for developing the early midline landmark, the primitive streak (PS). This study focused on the role of cell proliferation for midline development at pre- and post-PS-extension stages and analyzed PCP signaling components at post-PS-extension stages.</p><p><strong>Results: </strong>In contrast to pre-PS-extension stages, embryos under mitotic arrest during the post-PS-extension preserved notochord (NC) extension and Hensen's node (HN)/PS regression judged by both morphology and marker genes; although they became shorter, their lengths remained proportional to the embryo length. Laterality and segmentation of paraxial mesoderm were lost upon mitotic arrest. Accompanied by mitotic arrest-induced embryonic size reduction, cells including midline tissue displayed hypertrophy.</p><p><strong>Conclusion: </strong>This study has identified at least two distinct mitosis sensitivity phases during early midline development: One is PS extension that requires both mitosis and PCP, and the other is mitotic arrest-resistant midline development at post-PS-extension stages, with a still undefined influence by PCP signaling components.</p>","PeriodicalId":11247,"journal":{"name":"Developmental Dynamics","volume":" ","pages":"486-502"},"PeriodicalIF":1.5,"publicationDate":"2026-05-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12354007/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144274442","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Meet the editorial team: An interview with Maria A. Ros, Associate Editor, Developmental Dynamics, Instituto de Biomedicina y Biotecnología de Cantabria (ICSIC-UC), Santander, Spain.","authors":"Maria A Ros, Paul Trevorrow","doi":"10.1002/dvdy.70123","DOIUrl":"10.1002/dvdy.70123","url":null,"abstract":"","PeriodicalId":11247,"journal":{"name":"Developmental Dynamics","volume":" ","pages":"448-450"},"PeriodicalIF":1.5,"publicationDate":"2026-05-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146178361","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Differences in palatal shelf epithelial stiffness between the lingual/nasal and buccal/oral surfaces during palatal shelf elevation in developing mice.","authors":"Arata Nagasaka, Yasuhiko Bando, Miyuki Toda-Fujii, Go Onozawa, Kaito Suzuki, Takaki Miyata, Osamu Amano","doi":"10.1002/dvdy.70044","DOIUrl":"10.1002/dvdy.70044","url":null,"abstract":"<p><strong>Background: </strong>During secondary palate formation, bilateral palatal shelves grow vertically to a horizontal position. This morphological change of the palatal shelves, defined as the palatal shelf elevation, occurs from embryonic day (E)-13.5 to E14 in mice. Palatal shelves show regional differences in elevation patterns along the anterior-posterior (AP) axis; however, the underlying mechanisms remain unclear. Material properties of the lingual/nasal and buccal/oral surfaces, especially stiffness, possibly contribute to different elevation patterns.</p><p><strong>Results: </strong>Indentation test using atomic force microscopy was performed to measure the stiffness at the epithelial surface of the palatal shelf. Measurement of palatal shelf stiffness along the AP axis before and after elevation revealed that the lingual/nasal surface was softer than the buccal/oral surface in the posterior region before elevation and that the palatal shelf was stiffer after elevation than before elevation. Moreover, the thickness of epithelial cells on the lingual/nasal side was lower than that on the buccal/oral side before elevation.</p><p><strong>Conclusion: </strong>Overall, our results suggest that epithelial cell thickness affects epithelial surface stiffness, causing regional differences in elevation patterns.</p>","PeriodicalId":11247,"journal":{"name":"Developmental Dynamics","volume":" ","pages":"464-474"},"PeriodicalIF":1.5,"publicationDate":"2026-05-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144132167","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Kwan-Young Kim, You-Lim Hwang, Sunwoo Yeom, Seung-Hae Kwon, Sang-Hak Jeon
{"title":"Pss knockdown in the midgut causes growth retardation in Drosophila similar to that in human LMHD.","authors":"Kwan-Young Kim, You-Lim Hwang, Sunwoo Yeom, Seung-Hae Kwon, Sang-Hak Jeon","doi":"10.1002/dvdy.70039","DOIUrl":"10.1002/dvdy.70039","url":null,"abstract":"<p><strong>Background: </strong>Phosphatidylserine synthase (PSS), localized in the mitochondrial membrane, synthesizes phosphatidylserine. In humans, mutations in Pss lead to Lenz-Majewski hyperostotic dwarfism, a disorder affecting growth and development. The effects of Pss mutations on the growth of Drosophila melanogaster are not fully known. Hence, this study was conducted to investigate the effects of Pss knockdown on the growth and development of D. melanogaster.</p><p><strong>Results: </strong>Enterocyte (EC)-specific Pss knockdown resulted in reduced cell size in the gut via reduced Akt signaling. EC-specific Pss knockdown was associated with a decrease in gut size, a change in gut pH, and reduced food intake. These abnormalities affected normal nutrient metabolism in larvae, leading to decreased secretion of Drosophila insulin-like peptides. Consequently, the reduced systemic Akt signaling at the organismal level resulted not only in impaired gut growth but also in abnormal organismal growth and development.</p><p><strong>Conclusion: </strong>These findings highlight the significant role of the Pss gene in the growth and development of D. melanogaster.</p>","PeriodicalId":11247,"journal":{"name":"Developmental Dynamics","volume":" ","pages":"451-463"},"PeriodicalIF":1.5,"publicationDate":"2026-05-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13163622/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144118587","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Whole tissue imaging of cellular boundaries at sub-micron resolutions for deep learning cell segmentation: Applications in the analysis of epithelial bending of ectoderm.","authors":"Sam C P Norris, Jimmy K Hu, Neil H Shubin","doi":"10.1002/dvdy.70061","DOIUrl":"10.1002/dvdy.70061","url":null,"abstract":"<p><strong>Background: </strong>To understand cellular morphology, biologists have relied on traditional optical microscopy of tissues combined with tissue clearing protocols to image structures deep within tissues. Unfortunately, these protocols often struggle to retain cell boundary markers, especially at high enough resolutions necessary for precise cell segmentation. This limitation affects the ability to study changes in cell shape during major developmental events.</p><p><strong>Results: </strong>We introduce a method that preserves cell boundary markers and matches the refractive index of tissues with water. This technique enables the use of high-magnification, long working distance water-dipping objectives that provide sub-micron resolution images. We subsequently segment individual cells using a trained neural network segmentation model. These segmented images facilitate quantification of cell properties of the entire three-dimensional tissue. As a demonstration, we examine mandibles of transgenic mice that express fluorescent proteins in their cell membranes and extend this technique to a non-model animal, the catshark, investigating its dental lamina and dermal denticles-invaginating and evaginating ectodermal structures, respectively. This technique provides insight into the mechanical environment that cells experience during developmental transitions.</p><p><strong>Conclusions: </strong>This pipeline, named MORPHOVIEW, provides a powerful tool to quantify in high throughput the 3D structures of cells and tissues during organ morphogenesis.</p>","PeriodicalId":11247,"journal":{"name":"Developmental Dynamics","volume":" ","pages":"503-521"},"PeriodicalIF":1.5,"publicationDate":"2026-05-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12818342/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144728711","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Fumiya Meguro, Katsushige Kawasaki, Yoshito Kakihara, Maiko Kawasaki, Makoto Fukushima, Finsa Tisna Sari, Vanessa Utama, Alex Kesuma, Jun Nihara, Takehisa Kudo, Akira Fujita, Kaya Ichikawa, Kazuaki Osawa, Takeyasu Maeda, Koichi Tabeta, Makio Saeki, Atsushi Ohazama
{"title":"DNA repair during embryonic epidermal stratification.","authors":"Fumiya Meguro, Katsushige Kawasaki, Yoshito Kakihara, Maiko Kawasaki, Makoto Fukushima, Finsa Tisna Sari, Vanessa Utama, Alex Kesuma, Jun Nihara, Takehisa Kudo, Akira Fujita, Kaya Ichikawa, Kazuaki Osawa, Takeyasu Maeda, Koichi Tabeta, Makio Saeki, Atsushi Ohazama","doi":"10.1002/dvdy.70046","DOIUrl":"10.1002/dvdy.70046","url":null,"abstract":"<p><strong>Background: </strong>Genomes are constantly exposed to a myriad of DNA-damaging agents. Robust DNA repair mechanisms protect DNA by removing or tolerating damage. However, it remains unclear whether these mechanisms are required for organogenesis.</p><p><strong>Results: </strong>Multiple epithelial layers are essential for skin function, including body protection. The epidermis is initiated as a single layer and then stratifies in utero. Stratification did not occur in mice with epithelial conditional deletion of the DNA repair molecule Reptin (Reptin<sup>fl/fl</sup>;K14Cre). DNA damage was observed in the mutant epidermis but not in the wild-type epidermis. The mutant epidermis also showed reduced cell proliferation and upregulated p53 expression. Stratification was restored when p53 was deleted in the Reptin mutant mice by generating Reptin and p53 double mutant mice (Reptin<sup>fl/fl</sup>;K14Cre;p53<sup>-/-</sup>).</p><p><strong>Conclusion: </strong>In the wild-type epidermis, DNA is likely damaged at the initiation of embryonic stratification and promptly repaired by DNA repair mechanisms involving Reptin.</p>","PeriodicalId":11247,"journal":{"name":"Developmental Dynamics","volume":" ","pages":"475-485"},"PeriodicalIF":1.5,"publicationDate":"2026-05-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144172914","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Emerging therapeutic significance of MEF2C: Structure, function and its implications in diseases.","authors":"Jiawei Zhang, Yueyue Xie, Shuo Jiang, Shuo Huang, Hexuan Lv, Yuming Qin, Jinglei Xu, Zihan Dai, Haiqiang Wang, Yuanyuan Dai, Yutian Yang, Yang An, Guosen Zhang","doi":"10.1002/dvdy.70138","DOIUrl":"https://doi.org/10.1002/dvdy.70138","url":null,"abstract":"<p><p>Myocyte enhancer factor 2C (MEF2C) is a critical transcription factor and plays a pivotal role in muscle development, cardiac reprogramming, cellular differentiation, and disease pathogenesis. In this review, we have systematically described the structural characteristics of the MEF2C protein, particularly its DNA-binding MADS-box and transcriptional regulatory domains, and outlined the multi-level regulation mechanism of its expression and activity. We further have highlighted the functional roles of MEF2C in heart and skeletal muscle development, including its involvement in cardiomyocyte differentiation and reprogramming, and its pathological implications in heart and skeletal muscle diseases. Finally, we have summarized the pathological implications of MEF2C in neurological disorders and tumorigenesis and underlined its role as a regulator in the body's inflammatory response, offering a theoretical foundation for understanding its physiological and pathological implications and for the development of therapeutic strategies targeting MEF2C-related diseases.</p>","PeriodicalId":11247,"journal":{"name":"Developmental Dynamics","volume":" ","pages":""},"PeriodicalIF":1.5,"publicationDate":"2026-04-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147728752","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}