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Article
| Open Access
Combinatorial 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 Access
A 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 Access
GLI 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
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Article
| Open Access
Asymmetric requirement of Dpp/BMP morphogen dispersal in the Drosophila wing disc
Morphogens disperse to pattern tissues and control their growth during development, allowing for the specification of multiple fates across space. Here the authors block dispersal of a morphogen Dpp (BMP2/4) and show that the requirement for Dpp dispersal is much lower than previously thought.
- Shinya Matsuda
- , Jonas V. Schaefer
- & Markus Affolter
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Article
| Open Access
Self-organized formation of developing appendages from murine pluripotent stem cells
Organoid technology has been applied to model many types of organs in vitro, although structures containing multiple germ layers, such as limb buds, have not been generated. Here, the authors induce limb bud-like tissues from mouse ESCs that can be functionally integrated after transplantation.
- Shunsuke Mori
- , Eriko Sakakura
- & Mototsugu Eiraku
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Article
| Open Access
Evidence against tetrapod-wide digit identities and for a limited frame shift in bird wings
The homology of digits across amniotes is debated. Here, the authors compare the developmental transcriptomes of digits across five divergent amniotes and show high evolutionary dynamism in expression profiles, with conservation of a distinct developmental identity only in the anterior-most digit.
- Thomas A. Stewart
- , Cong Liang
- & Günter P. Wagner
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Article
| Open Access
Co-option of the cardiac transcription factor Nkx2.5 during development of the emu wing
The transcription factor Nkx2.5 is essential for heart development. Here, the authors identify a previously unknown expression domain for Nkx2.5 in the emu wing and explore its role in diminished wing bud development in the flightless emu, compared with three other birds that have functional wings.
- Peter G. Farlie
- , Nadia M. Davidson
- & Craig A. Smith
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| Open Access
Pervasive translational regulation of the cell signalling circuitry underlies mammalian development
Gene expression is regulated at several levels, including through the modulation of protein translation. Here the authors find that translation control diversifies gene expression between developing tissues and regulates major signalling pathways through a complex landscape of upstream open reading frames (uORFs).
- Kotaro Fujii
- , Zhen Shi
- & Maria Barna
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Article
| Open Access
JAK/STAT controls organ size and fate specification by regulating morphogen production and signalling
Morphogens regulate growth and pattern in vertebrate and invertebrate limbs. Here, the authors show that the JAK/STAT pathway promotes the growth of the Hedgehog-producing cell population in theDrosophilawing, thereby allowing the stable localization of the BMP/Dpp organizer.
- Carles Recasens-Alvarez
- , Ana Ferreira
- & Marco Milán
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Article
| Open Access
An orphan gene is necessary for preaxial digit formation during salamander limb development
The first digits to form in salamanders are the two most anterior ones, unlike in other tetrapods. Here, the authors show that the gene Prod1is expressed during early outgrowth of the limb bud and is necessary for limb formation in salamanders.
- Anoop Kumar
- , Phillip B. Gates
- & Jeremy P. Brockes
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Article
| Open Access
An intrinsic timer specifies distal structures of the vertebrate limb
The specification of positional values along the proximo-distal axis (shoulder to digits) of the vertebrate limb is an unresolved issue. By using heterochronic transplants of distal mesenchyme, the authors show that the zeugopod and autopod (elbow to digits) are progressively specified in an intrinsically timed manner.
- Patricia Saiz-Lopez
- , Kavitha Chinnaiya
- & Matthew Towers
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Article
| Open Access
Sonic hedgehog-expressing cells in the developing limb measure time by an intrinsic cell cycle clock
During vertebrate limb patterning the morphogen Sonic Hedgehog (Shh) is produced by cells of the polarizing region (ZPA). Here, the authors show, using chick embryo grafting experiments, that the duration of Shh expression by ZPA cells is defined by a cell cycle clock that is started and can also be reset by changes in retinoic acid signalling.
- Kavitha Chinnaiya
- , Cheryll Tickle
- & Matthew Towers
Suppression of apoptosis impairs phalangeal joint formation in the pathogenesis of brachydactyly type A1