ArticleJournal of pediatric surgery2013
Novel exomphalos genetic mouse model: the importance of accurate phenotypic classification.
Article in Journal of pediatric surgery, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 8 papers.
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Who cites it
8 citing papers in PubMed, 10 citations in OpenAlex.
- Ventral body wall closure: Mechanistic insights from mouse models and translation to human pathology.Developmental dynamics : an official publication of the American Association of Anatomists · 2025Review
- SCRIB controls apical contractility during epithelial differentiation.The Journal of cell biology · 2023Article
- Caldesmon ablation in mice causes umbilical herniation and alters contractility of fetal urinary bladder smooth muscle.The Journal of general physiology · 2021Article
- The Fgf8 subfamily (Fgf8, Fgf17 and Fgf18) is required for closure of the embryonic ventral body wall.Development (Cambridge, England) · 2020Article
- Transgelin-expressing myofibroblasts orchestrate ventral midline closure through TGFβ signalling.Development (Cambridge, England) · 2017Article
- Epiboly generates the epidermal basal monolayer and spreads the nascent mammalian skin to enclose the embryonic body.Journal of cell science · 2016Article
- Genetic interactions between planar cell polarity genes cause diverse neural tube defects in mice.Disease models & mechanisms · 2014Article
- Dermatan sulfate epimerase 1 deficient mice as a model for human abdominal wall defects.Birth defects research. Part A, Clinical and molecular teratology · 2014Article
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Authors and funding
13 authors at 1 institution in 1 country.
Funding
Abstract
backgroundRodent models of abdominal wall defects (AWD) may provide insight into the pathophysiology of these conditions including gut dysfunction in gastroschisis, or pulmonary hypoplasia in exomphalos. Previously, a Scribble mutant mouse model (circletail) was reported to exhibit gastroschisis. We further characterise this AWD in Scribble knockout mice.
methodHomozygous Scrib knockout mice were obtained from heterozygote matings. Fetuses were collected at E17.5-18.5 with intact amniotic membranes. Three mutants and two control fetuses were imaged by in amnio micro-MRI. Remaining fetuses were dissected, photographed and gut length/weight measured. Ileal specimens were stained for interstitial cells of Cajal (ICC), imaged using confocal microscopy and ICC quantified.
results127 fetuses were collected, 15 (12%) exhibited AWD. Microdissection revealed 3 mutants had characteristic exomphalos phenotype with membrane-covered gut/liver herniation into the umbilical cord. A further 12 exhibited extensive AWD, with eviscerated abdominal organs and thin covering membrane (intact or ruptured). Micro-MRI confirmed these phenotypes. Gut was shorter and heavier in AWD group compared to controls but morphology/number of ICC was not different. DISCUSSION: The Scribble knockout fetus exhibits exomphalos (intact and ruptured), in contrast to the original published phenotype of gastroschisis. Detailed dissection of fetuses is essential ensuring accurate phenotyping and result reporting.
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