Developmental Biology

Arrayed single-gene perturbations identify drivers of human anterior neural tube closure

1. Roya E Huang
2. Giridhar M Anand
3. Heitor C Megale
4. Jason Chen
5. Chudi Abraham-Igwe
6. Sharad Ramanathan

• Curated by eLife

  eLife Assessment

  This landmark study investigates how patterned human gastruloids can provide insights into neural tube closure. Using a screen, they identified positive and negative regulators and defines the epistasis among them using optimization of micro-pattern based gastruloid protocol and CRISPRi. This technical tour de force is exceptional and one of the first studies to reveal new knowledge on human development through embryo models.

Reviewed by eLife

Label-free assessment of pre-implantation embryo quality by the Fluorescence Lifetime Imaging Microscopy (FLIM)-phasor approach

1. Ning Ma
2. Nabora Reyes de Mochel
3. Paula Duyen Pham
4. Tae Yeon Yoo
5. Ken W. Y. Cho
6. Michelle A. Digman

Reviewed by Arcadia Science

Drosophila ryanodine receptor gene triggers functional and developmental muscle properties and could be used to assess the impact of human RYR1 mutations

1. Monika Zmojdzian
2. Teresa Jagla
3. Florian Cherik
4. Magda Dubinska-Magiera
5. Marta Migocka-Patrzalek
6. Malgorzata Daczewska
7. John Rendu
8. Krzysztof Jagla
9. Catherine Sarret

• Curated by eLife

  eLife Assessment

  This important paper provides novel information on the function of the Drosophila ryanodine receptor (RyR) during muscle development. The authors analyze the effects of a rare human mutation that causes myopathy that affects a conserved region of the gene. They present compelling evidence that this variant affects muscle function in flies. These results suggest that Drosophila can be used as a tool for screening additional variants.

  [Editors' note: this paper was reviewed by Review Commons.]

Reviewed by eLife

A cell atlas of the developing human outflow tract of the heart and its adult aortic valve derivatives

1. Rotem Leshem
2. Syed Murtuza Baker
3. Joshua Mallen
4. Lu Wang
5. John Dark
6. Andrew D Sharrocks
7. Karen Piper Hanley
8. Neil A Hanley
9. Magnus Rattray
10. Simon D Bamforth
11. Nicoletta Bobola

• Curated by eLife

  eLife Assessment

  This is a valuable study that presents human single nuclei RNA-seq and spatial transcriptomics data of the developing outflow tract and adult aortic valves that will facilitate research in this area. Data presented are solid, with bioinformatics analyses showing cell lineage and trajectory relationships, intriguingly suggesting persistence of embryonic signature in adult aortic valve cells. The latter results would be strengthened by experimental validation.

Reviewed by eLife

Inverted Assembly of the Lens Within Ocular Organoids Reveals Alternate Paths to Ocular Morphogenesis

1. Elin Stahl
2. Miguel Angel Delgado-Toscano
3. Ishwariya Saravanan
4. Anastasija Paneva
5. Joachim Wittbrodt
6. Lucie Zilova

• Curated by eLife

  eLife Assessment

  This important study demonstrates that ocular organoids can generate both retina and lens through a non-canonical, "inside-out" morphogenetic route. The work is supported by convincing data, with well-designed experiments combining imaging, molecular analysis, and transcriptomics to establish that lens formation in organoids follows conserved molecular programs despite an alternative morphogenesis. These findings expand our understanding of self-organization and developmental plasticity, and will be of broad interest to researchers working on eye development, organoids, and tissue engineering.

  [Editors' note: this paper was reviewed by Review Commons.]

Reviewed by eLife, Review Commons

Lineage priming and cell type proportioning depends on the interplay between stochastic and deterministic factors

1. William M Salvidge
2. Chris Brimson
3. Nicole Gruenheit
4. Li-Yao Huang
5. Catherine J Pears
6. Jason B Wolf
7. Christopher RL Thompson

• Curated by eLife

  eLife Assessment

  This important study shows how stochastic and deterministic factors are integrated in Dictyostelium discoideum to reliably drive determination of distinct cell types despite exposure to nearly identical environmental conditions. The authors present convincing evidence that gene expression variability contributes to the robustness of cell fate decisions, which reveals an unexpected role of stochasticity during cell differentiation.

Reviewed by eLife

In situ mutational screening and CRISPR interference define apterous cis-regulatory inputs during compartment boundary formation

1. Gustavo Aguilar
2. Michèle Sickmann
3. Dimitri Bieli
4. Gordian Born
5. Markus Affolter
6. Martin Müller

• Curated by eLife

  eLife Assessment

  This important paper presents the discovery of the molecular basis of differential apterous expression during early Drosophila wing disc development. The evidence supporting these conclusions is compelling, ranging from classical genetic approaches to state-of-the-art genetic engineering techniques. By opening new questions, this paper is expected to be of broad interest to developmental biologists and geneticists working on transcriptional regulation.

Reviewed by eLife

Mural cells protect the adult brain from hemorrhage but do not control the blood-brain barrier in developing zebrafish

1. Oguzhan F Baltaci
2. Andrea Usseglio Gaudi
3. Stefanie Dudczig
4. Weili Wang
5. Scott Paterson
6. Maria Cristina Rondon-Galeano
7. Ye-Wheen Lim
8. James Rae
9. Anne Lagendijk
10. Robert G Parton
11. Alison Farley
12. Benjamin M Hogan

• Curated by eLife

  eLife Assessment

  This important study addresses the contribution of pericytes to the organization and permeability control of the zebrafish blood-brain barrier (BBB). By analyzing pdgfrb mutant zebrafish that lack brain pericytes, the authors reveal that the resulting cerebrovascular network is abnormally patterned. Remarkably, however, the barrier retains its restrictive permeability during larval and juvenile stages. More pronounced vascular defects become evident in adults, where localized BBB leakage coincides with hemorrhages and aneurysm formation. Based on convincing and beautifully documented imaging data, the authors argue that, unlike what has been reported in rodent systems, pdgfrb-dependent pericytes are not essential for maintaining BBB integrity in the zebrafish brain.

Reviewed by eLife

Prickle and Ror modulate Dishevelled-Vangl interaction to regulate non-canonical Wnt signaling during convergent extension

1. Hwa-seon Seo
2. Deli Yu
3. Ivan Popov
4. Jiahui Tao
5. Allyson Angermeier
6. Fei Yang
7. Sylvie Marchetto
8. Jean-Paul Borg
9. Bingdong Sha
10. Jeffrey D Axelrod
11. Chenbei Chang
12. Jianbo Wang

• Curated by eLife

  eLife Assessment

  This valuable study addresses mechanisms of feedback inhibition between planar cell polarity protein complexes during convergent extension movements in Xenopus embryos. The authors propose a conceptually new model, in which non-canonical Wnt ligand stimulates transition of Dishevelled from its complex with Vangl to Frizzled, with essential roles of Prickle and Ror in this process. The main observations supporting molecular interactions rely on modest but significant changes in protein association in response to Wnt11. While the study is limited due to insufficient phenotypic analysis at the cellular level and the use of exogenously supplied proteins, this work is convincing and will be of broad interest to cell and developmental biologists.

Reviewed by eLife

A Novel 3D Visualization Method in Mice Identifies the Periportal Lamellar Complex (PLC) as a Key Regulator of Hepatic Ductal and Neuronal Branching Morphogenesis

1. Tongtong Xu
2. Fujun Cao
3. Ruihan Zhou
4. Qin Chen
5. Jian Zhong
6. Yulin Wang
7. Chaoxin Xiao
8. Banglei Yin
9. Chong Chen
10. Chengjian Zhao

• Curated by eLife

  eLife Assessment

  This study presents an important methodological advance-Liver-CUBIC combined with multicolor metallic nanoparticle perfusion-that enables high-resolution 3D visualization of the liver's complex multi-ductal architecture. The identification of the Periportal Lamellar Complex (PLC) as a novel perivascular structure with distinct cellular composition and low-permeability characteristics is convincing, supported by rigorous imaging data. The observed scaffolding role during fibrosis offers intriguing biological insights, though the functional claims would benefit from direct experimental validation.

Reviewed by eLife