Abstract：
:This study utilizes morphological and mechanistic endpoints to characterize the onset of bilateral atresia of the vas deferens in a recently derived cystic fibrosis (CF) rat model. Embryonic reproductive structures, including Wolffian (mesonephric) duct, Mullerian (paramesonephric) duct, mesonephric tubules, and gonad, were shown to mature normally through late embryogenesis, with involution of the vas deferens and/or epididymis typically occurring between birth and postnatal day 4 (P4), although timing and degree of atresia varied. No evidence of mucus obstruction, which is associated with pathology in other CF-affected tissues, was observed at any embryological or postnatal time point. Reduced epididymal coiling was noted post-partum and appeared to coincide with, or predate, loss of more distal vas deferens structure. Remarkably, α smooth muscle actin expression in cells surrounding duct epithelia was markedly diminished in CF animals by P2.5 when compared to wild type counterparts, indicating reduced muscle development. RNA-seq and immunohistochemical analysis of affected tissues showed disruption of developmental signaling by Wnt and related pathways. The findings have relevance to vas deferens loss in humans with CF, where timing of ductular damage is not well characterized and underlying mechanisms are not understood. If vas deferens atresia in humans begins in late gestation and continues through early postnatal life, emerging modulator therapies given perinatally might preserve and enhance integrity of the reproductive tract, which is otherwise absent or deficient in 97% of males with cystic fibrosis.
journal_name
Mech Devjournal_title
Mechanisms of developmentauthors
Plyler ZE,Birket SE,Schultz BD,Hong JS,Rowe SM,Petty CF,Crowley MR,Crossman DK,Schoeb TR,Sorscher EJdoi
10.1016/j.mod.2018.10.002subject
Has Abstractpub_date
2019-02-01 00:00:00pages
15-26eissn
0925-4773issn
1872-6356pii
S0925-4773(18)30136-9journal_volume
155pub_type
杂志文章abstract：:Homeodomain transcription factor CnNK-2 seems to play a major role in foot formation in Hydra. Recently, we reported in vitro evidence indicating that CnNK-2 has autoregulatory features and regulates expression of the morphogenetic peptide pedibin. We proposed that CnNK-2 and pedibin synergistically orchestrate foot d...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/j.mod.2005.04.010
更新日期：2005-09-01 00:00:00
abstract：:Adhesion differences are the main driver of cell sorting and related processes such as boundary formation or tissue positioning. In the early amphibian embryo, graded variations in cadherin density and localized expression of adhesion-modulating factors are associated with regional differences in adhesive properties i...
journal_title：Mechanisms of development
pub_type： 杂志文章,评审
doi：10.1016/j.mod.2016.09.003
更新日期：2017-04-01 00:00:00
abstract：:The recent determination and annotation of the entire euchromatic sequence of the Drosophila melanogaster genome predicted the existence of about 13600 different genes (Science 287 (2000) 2185; http://www.fruitfly.org/annot/index.html). In parallel, the Berkeley Drosophila Genome Project (BDGP) has undertaken systemat...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/s0925-4773(01)00566-4
更新日期：2002-01-01 00:00:00
abstract：:Nanos family members have been shown to act as translational repressors in the Drosophila and Caenorhabditis elegans germline, but direct evidence is missing for a similar function in vertebrates. Using a tethered function assay, we show that Xenopus Nanos1 is a translational repressor and that association with the RN...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/j.mod.2010.12.001
更新日期：2011-01-01 00:00:00
abstract：:Homeobox genes regulate multiple aspects of mouse development including the axial patterning in the central nervous system. Some of the more divergent members of this gene family are expressed in the anterior region of the central nervous system where the Hox genes are not expressed. We previously reported the isolati...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/0925-4773(94)90089-2
更新日期：1994-08-01 00:00:00
abstract：:Smad5, together with Smad1 and Smad8, have been implicated as downstream signal mediators for several bone morphogenetic proteins (BMPs). Recent studies have shown that primordial germ cells (PGCs) are absent or greatly reduced in Bmp4 or Bmp8b mutant mice. To define the role of Smad5 in PGC development, we examined P...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/s0925-4773(01)00367-7
更新日期：2001-06-01 00:00:00
abstract：:The transcription factor scleraxis (SCX) is expressed throughout tendon development and plays a key role in directing tendon wound healing. However, little is known regarding its role in fetal or young postnatal tendons, stages in development that are known for their enhanced regenerative capabilities. Here we used RN...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/j.mod.2020.103635
更新日期：2020-09-01 00:00:00
abstract：:Growth is confined within a size that is normal for each species, revealing that somehow an organism 'knows' when this size has been reached. Within a species, growth is also variable, but despite this, proportion and structure are maintained. Perhaps, the key element in the control of size is the control of cell numb...
journal_title：Mechanisms of development
pub_type： 杂志文章,评审
doi：10.1016/j.mod.2003.06.004
更新日期：2003-11-01 00:00:00
abstract：:We have identified a novel chicken gene, cMespo, which encodes a basic-helix-loop-helix (bHLH) protein with sequence homology to a subgroup of bHLH transcription factors that have been implicated in somitogenesis. cMespo transcripts are first found in the primitive streak of gastrulating chick embryos (HH stage 4) and...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/s0925-4773(00)00424-x
更新日期：2000-10-01 00:00:00
abstract：:Epimorphic regeneration is a process allowing the animal to regain its lost structure which depends on the resident pluripotent stem cells as well as de-differentiation of existing cells to form multi-potent stem cells. Many studies have been done to understand the appendage regeneration mechanism. The animal model us...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/j.mod.2018.02.001
更新日期：2018-04-01 00:00:00
abstract：:We describe the cloning, expression pattern and functional overexpression analysis of Xotx5b, a new member of the Otx gene family in Xenopus laevis. Early expression of Xotx5b resembles that of Xotx2, being detected in the organizer region at early gastrula stage, and, shortly after, also in anterior neuroectoderm. Du...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/s0925-4773(00)00367-1
更新日期：2000-08-01 00:00:00
abstract：:All body pigment cells in vertebrates are derived from the neural crest. In fish the neural crest can generate up to six different types of pigment cells, as well as various non-pigmented derivatives. In mouse and zebrafish, extensive collections of pigmentation mutants have enabled dissection of many aspects of pigme...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/j.mod.2004.01.004
更新日期：2004-07-01 00:00:00
abstract：:Splotch mice, which harbour mutations in the Pax3 gene, exhibit neural crest-related abnormalities including pigmentation defects, reduced or absent dorsal root ganglia and failure of cardiac outflow tract septation in homozygotes. Although splotch neural crest cells fail to colonise target tissues, they initiate migr...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/s0925-4773(97)00151-2
更新日期：1997-12-01 00:00:00
abstract：:The mouse and human Hox complex consists of 39 genes in four linkage groups (A-D). Although the structure and expression patterns of most of these genes have been reported, the 5' members of the Hox C linkage group have been only partially characterized. Herein we report the primary and genomic structure of the mouse ...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/s0925-4773(97)00182-2
更新日期：1998-01-01 00:00:00
abstract：:XLPOU91, a POU-homeobox gene is expressed in a narrow window during early Xenopus development. We show that ectopic expression of XLPOU91 RNA causes severe posterior truncations in embryos without inhibiting the formation of Spemann's organizer. Ectopic XLPOU91 expression also inhibits mesoderm induction by fibroblast...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/s0925-4773(98)00006-9
更新日期：1998-02-01 00:00:00
abstract：:In Drosophila melanogaster, the patterning of dorsal appendages on the eggshell is strictly controlled by EGFR signaling. However, the number of dorsal appendages is remarkably diverse among Drosophila species. For example, D. melanogaster and D. virilis have two and four dorsal appendages, respectively. Here we show ...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/s0925-4773(03)00164-3
更新日期：2003-08-01 00:00:00
abstract：:The Ras protein activates at least three different pathways during early development. Two of them regulate mesodermal gene expression and the third is thought to participate in the control of actin cytoskeleton dynamics via the Ral protein. From a yeast two-hybrid screen of a Xenopus maternal cDNA library, we identifi...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/j.mod.2004.07.008
更新日期：2004-12-01 00:00:00
abstract：:In many animals, germline progenitors are kept undifferentiated to give rise to germline stem cells (GSCs), enabling continuous production of gametes throughout animal life. In the Drosophila ovary, GSCs arise from a subset of primordial germ cells (PGCs) that stay undifferentiated even after gametogenesis has started...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/j.mod.2013.01.002
更新日期：2013-04-01 00:00:00
abstract：:Using in situ hybridization on whole-mounts and sections of mouse embryos we have visualized the pattern of expression for the Eph receptor ligand ephrin-A5. Non neuronal expression domains include the ectoderm of the branchial arches, the ectoderm and mesenchyme surrounding the dorsal root ganglia, the intervertebral...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/s0925-4773(00)00252-5
更新日期：2000-05-01 00:00:00
abstract：:SPOCK is a modular proteoglycan, with homology with proteins involved in cell adhesion processes and neurogenesis. We have previously shown that SPOCK transcripts predominate in the adult mouse brain. Here, we report its expression during mouse embryonic development by in situ hybridization, and immunocytochemistry. S...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/s0925-4773(99)00255-5
更新日期：2000-02-01 00:00:00
abstract：:The homeotic or Hox genes encode a network of conserved transcription factors which provide axial positional information and control segment morphology in development and evolution. During embryonic brain development of Drosophila, the Hox gene labial (lab) is essential for tritocerebral neuromere specification; lab l...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/j.mod.2005.09.004
更新日期：2006-01-01 00:00:00
abstract：:Myogenic regulatory factors (MRFs) are known to have essential roles in both the establishment and differentiation of the skeletal muscle cell lineage. MyoD is expressed early in the Xenopus mesoderm where it is present and active several hours before the activation of muscle differentiation genes. Previous studies in...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/j.mod.2017.05.002
更新日期：2017-08-01 00:00:00
abstract：:Xenopus embryos in solutions containing suramin show a dose-dependent decrease in the formation of dorsoanterior structures. Continuous treatment with 1 mM suramin produces embryos without mesodermal derivatives but with mesenchymal cells. Brief immersions of 20 min were used to determine the most sensitive stages and...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/0925-4773(94)90054-x
更新日期：1994-01-01 00:00:00
abstract：:Cell-cell interactions during embryogenesis have been shown to be important for establishing developmental fates in a number of organisms such as Drosophila and Caenorhabditis. One class of genes shown to mediate this process are receptor-protein tyrosine kinases (R-PTKs). To examine whether R-PTKs might participate i...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/0925-4773(94)90078-7
更新日期：1994-05-01 00:00:00
abstract：:We present a complete staging table of normal development for the lungless salamander, Hemidactylium scutatum (Caudata: Plethodontidae). Terrestrial egg clutches from naturally ovipositing females were collected and maintained at 15 °C in the laboratory. Observations, photographs, and time-lapse movies of embryos were...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/j.mod.2014.12.007
更新日期：2015-05-01 00:00:00
abstract：:Mutations in the CFTR gene cause cystic fibrosis (CF) with myocardial dysfunction. However, it remains unknown whether CF-related heart disease is a secondary effect of pulmonary disease, or an intrinsic primary defect in the heart. Here, we used zebrafish, which lack lung tissue, to investigate the role of CFTR in ca...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/j.mod.2020.103627
更新日期：2020-09-01 00:00:00
abstract：:Msg1 and Mrg1 are founding members of a gene family which exhibit distinct patterns of gene expression during mouse embryogenesis. Sequence analysis reveals that these genes are unlike any other gene identified to date, but they share two near-identical sequence domains. The Msg1 and Mrg1 expression profiles during ea...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/s0925-4773(98)00011-2
更新日期：1998-03-01 00:00:00
abstract：:A key role for phosphorylation of Smad2 by TGFβ superfamily ligands in the axial patterning of early embryos is well established. The regulation and role of Smad2 signaling in post-neurula embryonic patterning, however, is less well understood. While a variety of TGFβ superfamily ligands are implicated in various stag...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/j.mod.2010.08.004
更新日期：2010-09-01 00:00:00
abstract：:We have generated a null allele of the mouse Msx1 homeobox gene by insertion of an nlacZ reporter gene into its homeobox. The sensitivity of beta-galactosidase detection permitted us to reveal novel aspects of Msx1 gene expression in heterozygous embryos, in particular in ectoderm and mesoderm during gastrulation, and...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/s0925-4773(97)00065-8
更新日期：1997-07-01 00:00:00
abstract：:We have made a null mutation in the mouse activin beta B gene by deleting the portion of the gene encoding the proteolytic cleavage site and the majority of the coding region for the mature processed protein. Mice homozygous for this mutation complete embryogenesis and are completely viable. Approximately 40% of the h...
journal_title：Mechanisms of development
pub_type： 杂志文章
doi：10.1016/0925-4773(94)90094-9
更新日期：1994-07-01 00:00:00
