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| Open AccessParenchymal cues define Vegfa-driven venous angiogenesis by activating a sprouting competent venous endothelial subtype
Organs develop unique vascular architectures to support physiological functions. Here, authors show that organo-typical vascular networks may arise from specific parenchymal cues activating unique endothelial subtypes and angiogenic sprouting processes.
- Laetitia Préau
- , Anna Lischke
- & Ferdinand le Noble
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Article
| Open AccessDecoding spatiotemporal transcriptional dynamics and epithelial fibroblast crosstalk during gastroesophageal junction development through single cell analysis
Elucidating the gastroesophageal junction’s development is key to comprehending its disease susceptibility. Here, the authors mapped its development, uncovering cellular diversity and interaction dynamics using advanced spatiotemporal single-cell analysis.
- Naveen Kumar
- , Pon Ganish Prakash
- & Cindrilla Chumduri
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Article
| Open AccessSingle cell atlas of Xenoturbella bocki highlights limited cell-type complexity
Recent phylogenetic analyses have identified orphan clades, including Xenacoelomorphs, that can offer insights into bilaterian evolution. Here they generate a cell type atlas of Xenoturbella bockithat highlights cellular diversity in the nervous system and other tissues, reinforcing the idea of parallel evolution of cell types across animals.
- Helen E. Robertson
- , Arnau Sebé-Pedrós
- & Heather Marlow
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Article
| Open AccessSuppression of apoptosis impairs phalangeal joint formation in the pathogenesis of brachydactyly type A1
Authors explore the biological mechanisms underlying a missing digit joint in Brachydactyly type A1. They showed that excess Indian Hedgehog proteins suppress the apoptosis level needed for the progression of digit joint development in a mouse model.
- Adrian On Wah Leung
- , Andrew Chung Hin Poon
- & Danny Chan
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Article
| Open AccessKey homeobox transcription factors regulate the development of the firefly’s adult light organ and bioluminescence
Adult firefly light organs exhibit flashing signals important for courtship, though how these organs form during development is largely unknown. Here the authors demonstrate that homeobox transcription factors play a patterning role in the development of the adult light organs.
- Xinhua Fu
- & Xinlei Zhu
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Article
| Open AccessEndothelial cells regulate alveolar morphogenesis by constructing basement membranes acting as a scaffold for myofibroblasts
During alveologenesis myofibroblasts contractions at terminal sacs produce alveoli in the lungs. Here they show that endothelial cells promote myofibroblast-driven alveologenesis by forming basement membranes via Rap1-induced integrin β1 activation.
- Haruko Watanabe-Takano
- , Katsuhiro Kato
- & Shigetomo Fukuhara
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Article
| Open AccessIdentification of PCPE-2 as the endogenous specific inhibitor of human BMP-1/tolloid-like proteinases
Most proteases have their own endogenous, specific inhibitors which protect living organisms from the deleterious effects of excessive proteolytic activity. Here, authors identify PCPE-2 as a potent and specific inhibitor of BMP-1/tolloid-like proteases.
- Sandrine Vadon-Le Goff
- , Agnès Tessier
- & Catherine Moali
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Article
| Open AccessProenkephalin-A secreted by renal proximal tubules functions as a brake in kidney regeneration
The coordination of pro- and anti-regenerative factors is essential for organ regeneration. The authors show here that proenkephalin-A, secreted by proximal renal tubules in zebrafish, negatively regulates hydrogen peroxide production remodelling H3K4me3 in renal progenitor cells and controlling kidney regeneration strength.
- Chi Liu
- , Xiaoliang Liu
- & Jinghong Zhao
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Article
| Open AccessInterlocking of co-opted developmental gene networks in Drosophila and the evolution of pre-adaptive novelty
During evolution, genes can be recruited to new positions to perform novel functions. This study shows one such co-option event, where the reused gene networks are initially interlocked, so that any changes because of their function in one organ are mirrored in the other organs even if they provide no selective advantage, opening the potential for acquiring a novel function.
- Sara Molina-Gil
- , Sol Sotillos
- & James C.-G. Hombría
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Article
| Open AccessSinus venosus adaptation models prolonged cardiovascular disease and reveals insights into evolutionary transitions of the vertebrate heart
Nr2fs are conserved transcription factors that regulate atrial chamber and venous development. Here, the authors use adult zebrafish nr2f1a mutants to investigate compensatory remodeling of the inflow tract and hypotheses of cardiac evolution.
- Jacob T. Gafranek
- , Enrico D’Aniello
- & Joshua S. Waxman
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Article
| Open AccessDeciphering early human pancreas development at the single-cell level
Here, the authors revealed molecular heterogeneity, developmental trajectory and regulatory network of early human pancreas development, and depict the whole progression of pancreatic organogenesis during the first trimester at the single-cell level.
- Zhuo Ma
- , Xiaofei Zhang
- & Tao Xu
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Article
| Open AccessspinDrop: a droplet microfluidic platform to maximise single-cell sequencing information content
Droplet microfluidics enables high-throughput single-cell sequencing, but often with increased noise. Here the authors report spinDrop (sorting picoinjection inDrop) to increase gene detection and reduce noise; they use this to generate a high-quality molecular atlas of mouse brain development.
- Joachim De Jonghe
- , Tomasz S. Kaminski
- & Florian Hollfelder
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| Open AccessIntegrative analysis of transcriptome dynamics during human craniofacial development identifies candidate disease genes
Craniofacial disorders are among the most common congenital defects. Here, the authors examined the genetic causes of non-syndromic craniofacial disorders during human development through analysis of gene expression and epigenomics.
- Tara N. Yankee
- , Sungryong Oh
- & Justin Cotney
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Article
| Open AccessThree-dimensional molecular architecture of mouse organogenesis
Qu et al. present a detailed three-dimensional spatial transcriptome atlas of all major organs in the mouse embryo at E13.5, providing a better understanding of organ development and cellular interactions during mammalian development.
- Fangfang Qu
- , Wenjia Li
- & Guangdun Peng
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Article
| Open AccessOpposing gene regulatory programs governing myofiber development and maturation revealed at single nucleus resolution
Researchers used single-nucleus RNA and ATAC sequencing to create an atlas of skeletal muscle development. They identified the gene programs and transcription factors that control muscle fiber development and maturation.
- Matthieu Dos Santos
- , Akansha M. Shah
- & Eric N. Olson
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Article
| Open AccessLactate-dependent transcriptional regulation controls mammalian eye morphogenesis
Using a combination of eye organoids and mouse models the authors identify a bioenergetic independent role of lactate as a cell signaling molecule required during early stages of eye formation in mice.
- Nozomu Takata
- , Jason M. Miska
- & Guillermo Oliver
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Article
| Open AccessHedgehog signaling via its ligand DHH acts as cell fate determinant during skeletal muscle regeneration
Successful skeletal muscle regeneration relies on the interplay of multiple cell populations. Here, the authors describe how ciliary Hedgehog signaling coordinates the intercellular crosstalk required to balance wound healing and fatty fibrosis.
- Alessandra M. Norris
- , Ambili Bai Appu
- & Daniel Kopinke
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Article
| Open AccessInflationary theory of branching morphogenesis in the mouse salivary gland
The authors show that the ramified ductal network of the mouse salivary gland develops from a set of simple probabilistic rules, where ductal elongation and branching are driven by the persistent expansion of the surrounding tissue.
- Ignacio Bordeu
- , Lemonia Chatzeli
- & Benjamin D. Simons
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Article
| Open AccessM1BP is an essential transcriptional activator of oxidative metabolism during Drosophila development
The transcriptional regulation of mitochondrial oxidative phosphorylation gene expression is poorly understood. Using the developing Drosophila flight muscle, the authors identify the transcription factor M1BP as a new major regulator of this process.
- Gabriela Poliacikova
- , Marine Barthez
- & Andrew J. Saurin
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Article
| Open AccessMolecular mechanisms of tubulogenesis revealed in the sea star hydro-vascular organ
Understanding how cells organize into organs is a key effort in developmental biology. Here the authors introduce the sea star hydrovascular organ as a genetically tractable system to understand the contribution of cell migration and signaling pathways in tubulogenesis.
- Margherita Perillo
- , S. Zachary Swartz
- & Gary M. Wessel
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| Open AccessUSP7 controls NGN3 stability and pancreatic endocrine lineage development
Tightly controlled NGN3 expression is essential for endocrine cell generation in the developing pancreas, with dysregulation leading to hyperglycemia in mice. Here they identify USP7 as a key post-translational regulator of NGN3 stability and show that this axis is required for endocrine development and beta-cell differentiation.
- Teodora Manea
- , Jessica Kristine Nelson
- & Rocio Sancho
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Article
| Open AccessIsthmin-1 (Ism1) modulates renal branching morphogenesis and mesenchyme condensation during early kidney development
Loss of Ism1 in mice results in kidney agenesis and dysplasia that are common human diseases. Here they show that Ism1 is expressed in metanephric mesenchyme and acts as a ligand of Integrin α8β1 to regulate mesenchyme condensation during early renal branching morphogenesis.
- Ge Gao
- , Xiaoping Li
- & Zhongjun Zhou
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Article
| Open AccessEndothelial deletion of PTBP1 disrupts ventricular chamber development
Alternative splicing crucially affects various biological processes, however, its function in heart development is largely unknown. Here, the authors show an essential role of alternative splicing factor PTBP1 in ventricular chamber development.
- Hongyu Liu
- , Ran Duan
- & Yi-Han Chen
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Article
| Open AccessA defect in mitochondrial protein translation influences mitonuclear communication in the heart
The heart requires high levels of mitochondria to sustain function, and mitochondrial stressors can be transmitted to the nucleus and reprogram metabolism. Here, the authors show that a mitochondrial ribosomal protein is important for heart development in mice by increasing nuclear Klf15 expression.
- Feng Gao
- , Tian Liang
- & Jinghai Chen
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Article
| Open AccessVariations in the poly-histidine repeat motif of HOXA1 contribute to bicuspid aortic valve in mouse and zebrafish
Bicuspid aortic valve (BAV) is the most common cardiac defect and although highly heritable, few causal mutations have been identified. Here, the authors identify variants in the poly-histidine repeat motif of HOXA1 and show that its disruption leads to BAV in mice.
- Gaëlle Odelin
- , Adèle Faucherre
- & Stéphane Zaffran
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Article
| Open AccessCombinatorial effects on gene expression at the Lbx1/Fgf8 locus resolve split-hand/foot malformation type 3
Congenital limb defects are often associated with genomic rearrangements. Here they provide insights into the molecular mechanism underlying SHFM3-associated structural variations, offering a conceptual framework for how genomic rearrangements can alter gene expression and cause disease.
- Giulia Cova
- , Juliane Glaser
- & Stefan Mundlos
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Article
| Open AccessTesticular macrophages are recruited during a narrow fetal time window and promote organ-specific developmental functions
How testis resident macrophages develop and influence tissue function is not fully understood. Here the authors use mouse lineage tracing methods to document the haematopoietic source, development and recruitment of early testicular macrophages, support of foetal testis differentiation, and interaction with, and promotion of steroidogenesis in, Leydig cells.
- Xiaowei Gu
- , Anna Heinrich
- & Tony DeFalco
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Article
| Open AccessA single-cell transcriptional atlas reveals resident progenitor cell niche functions in TMJ disc development and injury
The transcriptional network in TMJ disc development and injury remains poorly characterized. Here they generate a scRNA-seq atlas of mouse TMJ disc, and identify the resident progenitor population and how its transcriptional reprogramming contributes to disc repair.
- Ruiye Bi
- , Qing Yin
- & Songsong Zhu
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Article
| Open AccessThe H2A.Z and NuRD associated protein HMG20A controls early head and heart developmental transcription programs
How the histone variant H2A.Z controls cell fate remains unclear. Here, the authors reveal that the H2A.Z interacting partner HMG20A plays a key role in regulating transcription during early head and heart development.
- Andreas Herchenröther
- , Stefanie Gossen
- & Sandra B. Hake
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Article
| Open AccessJag1-Notch cis-interaction determines cell fate segregation in pancreatic development
Notch signaling is crucial for pancreatic cell fate choice. With mathematical modeling and experiments, Xu et al. provides new insights into how different Notch ligands and Hes1 oscillation guide the spatial-temporal dynamics of cell differentiation.
- Xiaochan Xu
- , Philip Allan Seymour
- & Palle Serup
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| Open AccessMultiple pkd and piezo gene family members are required for atrioventricular valve formation
Cardiac valves are essential for heart function, and blood flow stimulation is critical for their formation. Here, researchers have identified a set of mechanosensory genes of the pkd and piezo families as key regulators of valve development.
- Thomas Juan
- , Agatha Ribeiro da Silva
- & Didier Y. R. Stainier
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Article
| Open AccessAdipose tissue is a source of regenerative cells that augment the repair of skeletal muscle after injury
The dynamics of fibroadipogenic progenitors (FAPs) after muscle injury are crucial to ensure efficient regeneration. Here the authors show that a pool of FAPs originates from adipose tissue and are necessary for effective muscle regeneration.
- Quentin Sastourné-Arrey
- , Maxime Mathieu
- & Coralie Sengenès
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Article
| Open AccessDOT1L regulates chamber-specific transcriptional networks during cardiogenesis and mediates postnatal cell cycle withdrawal
How and whether histone modifications regulate distinct gene networks remains insufficiently understood. Here Cattaneo et al show that DOT1L catalyzed H3K79me2 regulates fetal chamber-specific gene expression and neonatal cardiomyocyte cell cycle withdrawal to coordinate heart development.
- Paola Cattaneo
- , Michael G. B. Hayes
- & Sylvia M. Evans
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| Open AccessmTORC1 signaling facilitates differential stem cell differentiation to shape the developing murine lung and is associated with mitochondrial capacity
Lung branching requires differentiation of progenitor cells to be coordinated with morphogenetic events. Zhang et al. find that loss of mTORC1 signaling in the distal SOX9+ lung progenitors reduces mitochondrial capacity and ATP production, thus disrupting the formation of the conducting airways without affecting the development of the gas exchange unit.
- Kuan Zhang
- , Erica Yao
- & Pao-Tien Chuang
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| Open AccessHuman multilineage pro-epicardium/foregut organoids support the development of an epicardium/myocardium organoid
Stem cell models of organogenesis are a valuable tool for the study of human development, but often lack the context of tissue-tissue interaction. Here they generate human multi-lineage organoids comprising pro-epicardium, septum transversum, and liver bud, which they co-culture with heart organoids to generate a physiologically relevant model of organogenesis.
- Mariana A. Branco
- , Tiago P. Dias
- & Maria Margarida Diogo
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| Open AccessIdentification of evolutionarily conserved regulators of muscle mitochondrial network organization
Mitochondrial networks are carefully positioned to facilitate energy distribution within muscle cells. Here they show that energetic demands and conserved transcription factors regulate mitochondrial network organization and contractile phenotypes independently in Drosophila.
- Prasanna Katti
- , Peter T. Ajayi
- & Brian Glancy
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| Open AccessMitochondrial network configuration influences sarcomere and myosin filament structure in striated muscles
How different physical configurations between sarcomeres and mitochondria alter energetic support for contractile function of skeletal muscle is not clear. Here the authors use advanced 3D imaging and analysis techniques to show how space is made for mitochondria within the tightly packed sarcomere networks of striated muscle cells.
- Prasanna Katti
- , Alexander S. Hall
- & Brian Glancy
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| Open AccessA Dilp8-dependent time window ensures tissue size adjustment in Drosophila
Mechanisms ensuring developmental precision are poorly understood. Here Blanco-Obregon et al. report reciprocal feedback between Dilp8 and Ecdysone, two hormones required during a precise time window of Drosophila development for organ size adjustment.
- D. Blanco-Obregon
- , K. El Marzkioui
- & L. Boulan
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| Open AccessThe TFEB-TGIF1 axis regulates EMT in mouse epicardial cells
Epithelial-mesenchymal transition (EMT) is a complex process involved in organogenesis. Here, the authors show that the transcription factor EB (TFEB) regulates EMT in epicardium during heart development by tuning sensitivity to TGFβ signaling.
- Elena Astanina
- , Gabriella Doronzo
- & Federico Bussolino
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Article
| Open AccessA single WNT enhancer drives specification and regeneration of the Drosophila wing
The wing is a remarkable evolutionary novelty in insects. Here the authors demonstrate that the specification and regenerative capacity of the wing relies on a single wing-specific enhancer of the wingless gene in Drosophila.
- Elena Gracia-Latorre
- , Lidia Pérez
- & Marco Milán
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Article
| Open AccessN6-methyladenosine modification-mediated mRNA metabolism is essential for human pancreatic lineage specification and islet organogenesis
Ma et al. profile the dynamic landscape of m6A during pancreatic differentiation, and identify ALKBH5 as an essential m6A regulator supporting pancreatic differentiation, indicating a role for m6A-mediated mRNA metabolism in cell-fate specification.
- Xiaojie Ma
- , Jie Cao
- & Saiyong Zhu
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Article
| Open AccessMRTF specifies a muscle-like contractile module in Porifera
Myocytes are a key cell type that enable animal movement, but their evolutionary origins remain unclear. Colgren and Nichols describe molecular and functional similarities between a contractile module in tissues of a sponge and muscle tissues in other animals, indicating a common evolutionary origin.
- J. Colgren
- & S. A. Nichols
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Article
| Open AccessADAMTS18+ villus tip telocytes maintain a polarized VEGFA signaling domain and fenestrations in nutrient-absorbing intestinal blood vessels
The molecular mechanisms ensuring the specialized structure of small intestinal villus tip blood vessels are incompletely understood. Here the authors show that ADAMTS18+ telocytes maintain a “just-right” level and location of VEGFA signaling on intestinal villus blood vessels, thereby ensuring the presence of endothelial fenestrae for nutrient absorption, while avoiding excessive leakiness and destabilization of villus tip epithelial structures.
- Jeremiah Bernier-Latmani
- , Cristina Mauri
- & Tatiana V. Petrova
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Article
| Open AccessControl of CRK-RAC1 activity by the miR-1/206/133 miRNA family is essential for neuromuscular junction function
The miR-1/133/206 gene family codes for the most abundant microRNAs in striated muscles. Here, Klockner et al show that inactivation of all family members in skeletal muscle prevents formation of normal neuromuscular junctions due to increased expression of the adaptor protein CRK.
- Ina Klockner
- , Christian Schutt
- & Thomas Braun
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Article
| Open AccessSingle-cell chromatin profiling of the primitive gut tube reveals regulatory dynamics underlying lineage fate decisions
The primitive gut tube gives rise to all major internal organs, while underlying regulatory mechanisms are unclear. Here, the authors analyze its chromatin landscape at the single-cell level and define the epigenetic regulation of lineage fate decisions and plasticity in organ development and homeostasis.
- Ryan J. Smith
- , Hongpan Zhang
- & Tae-Hee Kim
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Article
| Open AccessMetabolic control of progenitor cell propagation during Drosophila tracheal remodeling
Tracheal remodeling is a key step during Drosophila metamorphosis. Here they report that tracheal progenitor cells are activated through Yorkie-dependent proliferation and migration, which is induced by metabolic deficit and insulin signaling.
- Yue Li
- , Pengzhen Dong
- & Hai Huang
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Article
| Open AccessTranscription factors AP-2α and AP-2β regulate distinct segments of the distal nephron in the mammalian kidney
How the distal nephron is patterned during kidney development has been difficult to study. Here they show that AP-2β is required for the formation and postnatal function of distal convoluted tubules, whereas AP-2α has a role in maintaining the structure of medullary collecting ducts.
- Joseph O. Lamontagne
- , Hui Zhang
- & Alexander G. Marneros
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Article
| Open AccessHand2 delineates mesothelium progenitors and is reactivated in mesothelioma
The mesothelium supports homeostasis and regeneration, yet its development origins remain unclear. Here, the authors uncovered the earliest mesothelium progenitor cells in zebrafish, linking Hand2 gene function to mesothelium formation and its re-activation to mesothelioma tumors.
- Karin D. Prummel
- , Helena L. Crowell
- & Christian Mosimann
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Article
| Open AccessGLI transcriptional repression is inert prior to Hedgehog pathway activation
GLI repression has been presumed to be the default transcriptional state and important for pre-patterning tissues. Challenging current models, the authors show that GLI3 repression is inert in the limb bud before the onset of Hedgehog signaling.
- Rachel K. Lex
- , Weiqiang Zhou
- & Steven A. Vokes