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Genesis: the Journal of Genetics and Development

Natalie Shukrun, Yehuda Shabtai, Graciela Pillemer, Abraham Fainsod
Intrauterine Growth Restriction (IUGR) is commonly observed in human pregnancies and can result in severe clinical outcomes. IUGR is observed in Fetal Alcohol Syndrome (FAS) fetuses as a result of alcohol (ethanol) exposure during pregnancy. To further understand FAS, the severe form of Fetal Alcohol Spectrum Disorder, we performed an extensive quantitative analysis of the effects of ethanol on embryo size utilizing our Xenopus model. Ethanol-treated embryos exhibited size reduction along the anterior-posterior axis...
January 23, 2019: Genesis: the Journal of Genetics and Development
Leah L Zagore, Cydni C Akesson, Donny D Licatalosi
Spermatogenesis is a highly ordered developmental program that produces haploid male germ cells. The study of male germ cell development in the mouse has provided unique perspectives into the molecular mechanisms that control cell development and differentiation in mammals, including tissue-specific gene regulatory programs. An intrinsic challenge in spermatogenesis research is the heterogeneity of germ and somatic cell types present in the testis. Techniques to separate and isolate distinct mouse spermatogenic cell types have great potential to shed light on molecular mechanisms controlling mammalian cell development, while also providing new insights into cellular events important for human reproductive health...
January 21, 2019: Genesis: the Journal of Genetics and Development
Sonia Sudiwala, Sarah M Knox
Organs and structures of the vertebrate head perform a plethora of tasks including visualization, digestion, vocalization/communication, auditory functions, and respiration in response to neuronal input. This input is primarily derived from afferent and efferent fibers of the cranial nerves (sensory and motor respectively) and efferent fibers of the cervical sympathetic trunk. Despite their essential contribution to the function and integration of processes necessary for survival, how organ innervation is established remains poorly understood...
January 9, 2019: Genesis: the Journal of Genetics and Development
Lan Hai, Maria M Szwarc, Denise G Lanza, Jason D Heaney, John P Lydon
The promyelocytic leukemia zinc finger (PLZF) transcription factor mediates a wide-range of biological processes. Accordingly, perturbation of PLZF function results in a myriad of physiologic defects, the most conspicuous of which is abnormal skeletal patterning. Although whole body knockout of Plzf in the mouse (PlzfKO ) has significantly expanded our understanding of Plzf function in vivo, a conditional knockout mouse model that enables tissue or cell-type specific ablation of Plzf has not been developed...
January 9, 2019: Genesis: the Journal of Genetics and Development
Josefina L Iungman, María N Molinero, Melina S Simoncini, Carlos I Piña
Here we describe the embryonic development of Salvator merianae external morphologic features, as based on observation of 28 embryos across different days of incubation at 31 ± 0.5°C. Observed developmental stages were grouped and classified into early, middle, and late periods. The early period (stages 3 to 11) is distinguished by the origin of the encephalic vesicles, sensory placodes, pharyngeal arches, and degree of body flexion and rotation. The medium period (stages 8 to 15) is distinguished by limb differentiation and by cranium-facial characteristics...
January 8, 2019: Genesis: the Journal of Genetics and Development
Gregg DiNuoscio, Radhika P Atit
Cranial neural crest cells (CNCC) give rise to cranial mesenchyme (CM) that differentiates into the forebrain meningeal progenitors in the basolateral and apical regions of the head. This occurs in close proximity to the other CNCC-CM-derivatives such as calvarial bone and and dermal progenitors. We found active Wnt signaling transduction in the forebrain meningeal progenitors in basolateral and apical populations and in the non-meningeal CM preceding meningeal differentiation. Here, we dissect the source of Wnt ligand secretion and requirement of Wnt/β-catenin signaling for the lineage selection and early differentiation of the forebrain meninges...
January 7, 2019: Genesis: the Journal of Genetics and Development
Berardino Petrelli, Liat Bendelac, Geoffrey G Hicks, Abraham Fainsod
Fetal Alcohol Spectrum Disorder (FASD) is a set of neurodevelopmental malformations caused by maternal consumption of alcohol during pregnancy. FASD sentinel facial features are unique to the disorder and microcephaly is common in severe forms of FASD. Retinoic acid deficiency has been shown to cause craniofacial malformations and microcephaly in animal models reminiscent of those caused by prenatal alcohol exposure. Alcohol exposure affects the migration and survival of cranial neural crest cells, which are required for proper frontonasal prominence and pharyngeal arch development...
January 7, 2019: Genesis: the Journal of Genetics and Development
Hiroshi Kiyonari, Mari Kaneko, Takaya Abe, Go Shioi, Shinichi Aizawa, Yasuhide Furuta, Toshihiko Fujimori
Live imaging is one of the most powerful technologies for studying the behaviors of cells and molecules in living embryos. Previously, we established a series of reporter mouse lines in which specific organelles are labeled with various fluorescent proteins. In this study, we examined the localizations of fluorescent signals during pre-implantation development of these mouse lines, as well as a newly established one, by time-lapse imaging. Each organelle was specifically marked with fluorescent fusion proteins; fluorescent signals were clearly visible during the whole period of time-lapse observation, and the expression of the reporters did not affect embryonic development...
December 29, 2018: Genesis: the Journal of Genetics and Development
Maneeshi S Prasad, Rebekah M Charney, Martín I García-Castro
The neural crest is a fascinating embryonic population unique to vertebrates that is endowed with remarkable differentiation capacity. Thought to originate from ectodermal tissue, neural crest cells generate neurons and glia of the peripheral nervous system, and melanocytes throughout the body. However, the neural crest also generates many ectomesenchymal derivatives in the cranial region, including cell types considered to be of mesodermal origin such as cartilage, bone, and adipose tissue. These ectomesenchymal derivatives play a critical role in the formation of the vertebrate head, and are thought to be a key attribute at the center of vertebrate evolution and diversity...
December 21, 2018: Genesis: the Journal of Genetics and Development
Praveer Sharma, Adam L MacLean, Lina Meinecke, David Clouthier, Qing Nie, Thomas F Schilling
The mandibular or first pharyngeal arch forms the upper and lower jaws in all gnathostomes. A gene regulatory network that defines ventral, intermediate, and dorsal domains along the dorsal-ventral (D-V) axis of the arch has emerged from studies in zebrafish and mice, but the temporal dynamics of this process remain unclear. To define cell fate trajectories in the arches we have performed quantitative gene expression analyses of D-V patterning genes in pharyngeal arch primordia in zebrafish and mice. Using NanoString technology to measure transcript numbers per cell directly we show that, in many cases, genes expressed in similar D-V domains and induced by similar signals vary dramatically in their temporal profiles...
December 18, 2018: Genesis: the Journal of Genetics and Development
Deborah L Chapman
Intermediate mesoderm (IM) is the strip of tissue lying between the paraxial mesoderm (PAM) and the lateral plate mesoderm that gives rise to the kidneys and gonads. Chick fate mapping studies suggest that IM is specified shortly after cells leave the primitive streak and that these cells do not require external signals to express IM-specific genes (James and Schultheiss, 2003). Surgical manipulations of the chick embryo, however, revealed that PAM-specific signals are required for IM differentiation into pronephros - the first kidney (Mauch et al...
December 13, 2018: Genesis: the Journal of Genetics and Development
Abigail A Kindberg, Jeffrey O Bush
Craniofacial morphogenesis is a highly dynamic process that requires changes in the behaviors and physical properties of cells in order to achieve the proper organization of different craniofacial structures. Boundary formation is a critical process in cellular organization, patterning, and ultimately tissue separation. There are several recurring cellular mechanisms through which boundary formation and cellular organization occur including, transcriptional patterning, cell segregation, cell adhesion and migratory guidance...
December 13, 2018: Genesis: the Journal of Genetics and Development
Elena V Fedorova, Natalja V Dorogova, Elena U Bolobolova, Svetlana A Fedorova, Dmitry A Karagodin, Anna A Ogienko, Asja S Khruscheva, Elina M Baricheva
Investigation of Drosophila oogenesis provides the opportunity to understand conservative genetic mechanisms underlying fertile female gamete development. In this work, we showed that the Drosophila DNA-binding protein GAGA factor (GAF) had a multifunctional role in oogenesis and it is involved in the regulation of this process genetic program. We studied the influence on Drosophila oogenesis of a number of mutations in the 5' region of the Trl gene that encodes GAF. We found that our originally generated Trl mutations lead to a decrease in transcriptional gene activity and levels of GAF expression in both germline and follicular cells...
December 10, 2018: Genesis: the Journal of Genetics and Development
Jeane Silva, Haiyan Qin, John K Cowell
Constitutional mutations in LGI1 predispose to an autosomal dominant epilepsy syndrome in humans and germline inactivation of Lgi1 in mice leads to early onset seizures. LGI1 is highly expressed in the regions involved in neuronal stem cell generation and migration and detailed analysis of the brains in these mice reveals a subtle cortical dysplasia characterized by hypercellularity in the outer cortical layers. To investigate the cellular origin for this cortical dysplasia, we created mice that allow cell-specific, conditional inactivation of Lgi1...
November 29, 2018: Genesis: the Journal of Genetics and Development
Shaughna Langerak, Alicia Trombley, Joseph R Patterson, Devon Leroux, Alexandra Couch, Megan P Wood, Jennifer A Schisa
A key aspect of development in all metazoans is remodeling at the cellular level. During the development of gametes, remodeling occurs throughout the germ line. When C. elegans hermaphrodites become depleted of sperm after 4 days of adulthood, significant cellular remodeling occurs within the meiotically-arrested oocytes, including the formation of ribonucleoprotein (RNP) granules. Since major remodeling of the endoplasmic reticulum (ER) occurs in early embryos, we investigated the extent of ER remodeling in meiotically-arrested oocytes...
November 29, 2018: Genesis: the Journal of Genetics and Development
Chyong-Yi Wu, Lisa A Taneyhill
The cranial trigeminal ganglia play a vital role in the peripheral nervous system through their relay of sensory information from the vertebrate head to the brain. These ganglia are generated from the intermixing and coalescence of two distinct cell populations: cranial neural crest cells and placodal neurons. Trigeminal ganglion assembly requires the formation of cadherin-based adherens junctions within the neural crest cell and placodal neuron populations; however, the molecular composition of these adherens junctions is still unknown...
November 21, 2018: Genesis: the Journal of Genetics and Development
Andrew J Conith, Daniel T Lam, R Craig Albertson
The shape of the craniofacial skeleton is constantly changing through ontogeny, and reflects a balance between developmental patterning and mechanical-load induced remodeling. Muscles are a major contributor to producing the mechanical environment that is crucial for "normal" skull development. Here we use an F5 hybrid population of Lake Malawi cichlids to characterize the strength and types of associations between craniofacial bones and muscles. We focus on four bones/bone complexes, with different developmental origins, alongside four muscles with distinct functions...
November 12, 2018: Genesis: the Journal of Genetics and Development
Ye Wang, Hejie Song, Wenfeng Wang, Zhen Zhang
Megf6, a member of MEGF (multiple EGF-like domains) protein family, is a conserved high molecular weight protein with 30 EGF-like domains. Although many members of the MEGF protein family are essential for embryonic development and homeostasis, the role of Megf6 in development and physiology is still unknown. Here, we generated Megf6-deficient mice using CRISPR-Cas9 technique and showed that Megf6 is dispensable for embryonic development. We also constructed the Megf6Cre allele to study Megf6-expressing cell lineages...
October 31, 2018: Genesis: the Journal of Genetics and Development
Shun-Wa Tsang, Yanjiang Guo, Long-Hei Chan, Yingyu Huang, King L Chow
MAB21L2(R51C) is one of the five documented MAB21L2 mutations in human patients with bilateral eye malformations identified via whole exome sequencing. In addition to the eye abnormality, patients with MAB21L2R51C/+ mutation also have skeletal dysplasia and intellectual disability. To evaluate the pathology of this mutant allele systematically in understanding the functional role of MAB21L2 in human development, we introduce the R51C mutation into the mouse genome by CRISPR/Cas9 system to generate a mouse model for detailed characterization...
October 30, 2018: Genesis: the Journal of Genetics and Development
C J Pickett, Robert W Zeller
Eliminating or silencing a gene's level of activity is one of the classic approaches developmental biologists employ to determine a gene's function. A recently developed method of gene perturbation called CRISPR-Cas, which was derived from a prokaryotic adaptive immune system, has been adapted for use in eukaryotic cells. This technology has been established in several model organisms as a powerful and efficient tool for knocking out or knocking down the function of a gene of interest. It has been recently shown that CRISPR-Cas functions with fidelity and efficiency in Ciona robusta...
October 30, 2018: Genesis: the Journal of Genetics and Development
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