Mouse Pitx2 deficiency leads to anomalies of the ventral body wall, heart, extra- and periocular mesoderm and right pulmonary isomerism

Development ◽  
1999 ◽  
Vol 126 (24) ◽  
pp. 5749-5758 ◽  
Author(s):  
K. Kitamura ◽  
H. Miura ◽  
S. Miyagawa-Tomita ◽  
M. Yanazawa ◽  
Y. Katoh-Fukui ◽  
...  

Pitx2, a bicoid-related homeobox gene, is involved in Rieger's syndrome and the left-right (L-R) asymmetrical pattern formation in body plan. In order to define the genomic structure and roles of Pitx2, we analyzed the genomic structure and generated Pitx2-deficient mice with the lacZ gene in the homeobox-containing exon of Pitx2. We were able to show that among three isoforms of Pitx2, Pitx2c shows asymmetrical expression whereas Pitx2a, Pitx2b and Pitx2c show symmetrical expression. In Pitx2(−)(/)(−) embryos there was an increase in mesodermal cells in the distal end of the left lateral body wall and an amnion continuous with the lateral body wall thickened in its mesodermal layer. These changes resulted in a failure of ventral body wall closure. In lung and heart in which Pitx2 is expressed asymmetrically, right pulmonary isomerism, atrioventricular canals with prominent swelling, and juxtaposition of the atrium were detected. The hearts failed to develop tricuspid and mitral valves and a common atrioventricular valve forms. Further, dysgenesis of the Pitx2(−)(/)(−) extraocular muscle and thickening of the mesothelial layer of cornea were observed in the ocular system where Pitx2 is expressed symmetrically, and these resulted in enophthalmos. The present study shows that Pitx2 expressed in various sites participates in morphogenesis through three types of actions: the involvement of asymmetric Pitx2 expression in the entire morphogenetic process of L-R asymmetric organs; the involvement of asymmetric Pitx2 expression in the regional morphogenesis of asymmetric organs; and finally the involvement of symmetric Pitx2 expression in the regional morphogenesis of symmetric organs.

2021 ◽  
Vol 9 (8) ◽  
pp. 848
Author(s):  
Elise E. B. LaDouceur ◽  
Linda A. Kuhnz ◽  
Christina Biggs ◽  
Alicia Bitondo ◽  
Megan Olhasso ◽  
...  

Sea pigs (Scotoplanes spp.) are deep-sea dwelling sea cucumbers of the phylum Echinodermata, class Holothuroidea, and order Elasipodida. Few reports are available on the microscopic anatomy of these deep-sea animals. This study describes the histologic findings of two, wild, male and female Scotoplanes sp. collected from Monterey Bay, California. Microscopic findings were similar to other holothuroids, with a few notable exceptions. Sea pigs were bilaterally symmetrical with six pairs of greatly enlarged tube feet arising from the lateral body wall and oriented ventrally for walking. Neither a rete mirabile nor respiratory tree was identified, and the large tube feet may function in respiration. Dorsal papillae protrude from the bivium and are histologically similar to tube feet with a large, muscular water vascular canal in the center. There were 10 buccal tentacles, the epidermis of which was highly folded. Only a single gonad was present in each animal; both male and female had histologic evidence of active gametogenesis. In the male, a presumed protozoal cyst was identified in the aboral intestinal mucosa, and was histologically similar to previous reports of coccidians. This work provides control histology for future investigations of sea pigs and related animals using bright field microscopy.


Development ◽  
1991 ◽  
Vol 112 (3) ◽  
pp. 807-811 ◽  
Author(s):  
K. Schughart ◽  
C.J. Bieberich ◽  
R. Eid ◽  
F.H. Ruddle

To characterize cis-acting regulatory elements of the murine homeobox gene, Hox-2.2, transgenic mouse lines were generated that contained the LacZ reporter gene under the control of different fragments from the presumptive Hox-2.2 promoter. A promoter region of 3600 base pairs (bp) was identified, which reproducibly directed reporter gene expression into specific regions of developing mouse embryos. At 8.5 days postcoitum (p.c.) reporter gene activity was detected in posterior regions of the lateral mesoderm and, in subsequent developmental stages, expression of the LacZ gene was restricted to specific regions of the developing limb buds and the mesenchyme of the ventrolateral body region. This pattern of Hox-2.2-LacZ expression was found in all transgenic embryos that have been generated with the 3.6 kb promoter fragment (two founder embryos and embryos from five transgenic lines). In addition, embryos from two transgenic mouse lines expressed the reporter gene at low levels in the developing central nervous system (CNS). Our results are consistent with the idea that in addition to their presumptive role in CNS and vertebrae development, Hox-2.2 gene products are involved in controlling pattern formation in developing limbs.


Development ◽  
1998 ◽  
Vol 125 (7) ◽  
pp. 1217-1227 ◽  
Author(s):  
B.T. Kehl ◽  
K.O. Cho ◽  
K.W. Choi

The Drosophila notum, the dorsal body wall of the thorax, is subdivided genetically into longitudinal domains (Calleja, M., Moreno, E., Pelaz, S. and Morata, G. (1996) Science 274, 252–255). Two homeobox genes clustered in the iroquois complex, araucan and caupolican, regulate proneural genes and are required for development of sensory bristles in the lateral notum (Gomez-Skarmeta, J. L., del Corral, R. D., de la Calle-Mustienes, E., Ferres-Marco, D. and Modolell, J. (1996) Cell 85, 95–105). An iroquois-related homeobox gene, mirror, was recently isolated and is localized close to the iroquois complex region (McNeil, H., Yang, C.-H., Brodsky, M., Ungos, J. and Simon, M. A. (1997) Genes and Development 11, 1073–1082; this study). We show that mirror is required for the formation of the alula and a subset of sensory bristles in the lateral domain of the notum. Genetic analysis suggests that mirror and the other iroquois genes interact to form the alula as well as the sensory organs. Based on similarities between mirror and the iroquois genes in their genetic map positions, expression, protein structure and function, mirror is considered a new member of the iroquois complex and is involved in prepatterning sensory precursor cells in the lateral notum.


2018 ◽  
Vol 32 (4) ◽  
pp. 328 ◽  
Author(s):  
Kailey Anderson ◽  
João Brandão ◽  
Christoph Mans
Keyword(s):  

1998 ◽  
Vol 9 (1) ◽  
pp. 81-83 ◽  
Author(s):  
Tetsuya A. Fujii ◽  
Kveta Cvecklova ◽  
Debra J. Gilbert ◽  
Neal G. Copeland ◽  
Nancy A. Jenkins ◽  
...  

2005 ◽  
Vol 168 (6) ◽  
pp. 941-953 ◽  
Author(s):  
Yoshihiko Shimizu ◽  
Dean Thumkeo ◽  
Jeongsin Keel ◽  
Toshimasa Ishizaki ◽  
Hiroko Oshima ◽  
...  

Rho-associated kinase (ROCK) I mediates signaling from Rho to the actin cytoskeleton. To investigate the in vivo functions of ROCK-I, we generated ROCK-I–deficient mice. Loss of ROCK-I resulted in failure of eyelid closure and closure of the ventral body wall, which gave rise to the eyes open at birth and omphalocele phenotypes in neonates. Most ROCK-I−/− mice died soon after birth as a result of cannibalization of the omphalocele by the mother. Actin cables that encircle the eye in the epithelial cells of the eyelid were disorganized and accumulation of filamentous actin at the umbilical ring was impaired, with loss of phosphorylation of the myosin regulatory light chain (MLC) at both sites, in ROCK-I−/− embryos. Stress fiber formation and MLC phosphorylation induced by EGF were also attenuated in primary keratinocytes from ROCK-I−/− mice. These results suggest that ROCK-I regulates closure of the eyelids and ventral body wall through organization of actomyosin bundles.


1994 ◽  
Vol 195 (1) ◽  
pp. 329-343 ◽  
Author(s):  
G Ehret ◽  
E Keilwerth ◽  
T Kamada

Frequency-response curves of the tympanum and lateral body wall (lung area) were measured by laser Doppler vibrometry in three treefrog (Smilisca baudini, Hyla cinerea, Osteopilus septentrionalis) and four dendrobatid frog (Dendrobates tinctorius, D. histrionicus, Epipedobates tricolor, E. azureiventris) species. The high-frequency cut-off of the body wall response was always lower than that of the tympanum. The best response frequencies of the lateral body wall were lower than those of the tympanum in some species (S. baudini, O. septentrionalis, D. tinctorius), while in the others they were rather similar. Best tympanic frequencies and best body wall response frequencies tended to differ more with increasing body size. Stimulation of the tympanum by sound transfer through 3.14 mm2 areas of the lateral body wall showed that the lung-eardrum pathway can be in two states, depending on breathing activity within the lungs: 44% (in Smilisca), 39% (in Hyla) and 31% (in Osteopilus) of the eardrum vibrations were 2.5-8 times (8-18 dB) larger when the frogs were breathing with the lungs compared with non-breathing conditions. The vibration amplitudes of the tympanum and lateral body wall of the treefrogs followed the same dependence on sound intensity, only absolute amplitudes differed between species. Our results suggest that the lung-eardrum pathway attenuates high-frequency components of species-specific calls and enhances low-frequency components. In addition, an amplitude modulation is imposed on the low frequencies during the rhythm of breathing.


Development ◽  
1999 ◽  
Vol 126 (6) ◽  
pp. 1225-1234 ◽  
Author(s):  
M. Campione ◽  
H. Steinbeisser ◽  
A. Schweickert ◽  
K. Deissler ◽  
F. van Bebber ◽  
...  

Left-right asymmetry in vertebrates is controlled by activities emanating from the left lateral plate. How these signals get transmitted to the forming organs is not known. A candidate mediator in mouse, frog and zebrafish embryos is the homeobox gene Pitx2. It is asymmetrically expressed in the left lateral plate mesoderm, tubular heart and early gut tube. Localized Pitx2 expression continues when these organs undergo asymmetric looping morphogenesis. Ectopic expression of Xnr1 in the right lateral plate induces Pitx2 transcription in Xenopus. Misexpression of Pitx2 affects situs and morphology of organs. These experiments suggest a role for Pitx2 in promoting looping of the linear heart and gut.


Development ◽  
1993 ◽  
Vol 117 (2) ◽  
pp. 769-778 ◽  
Author(s):  
S.J. Gaunt ◽  
M. Blum ◽  
E.M. De Robertis

After an earlier, transient phase of expression in the developing primitive streak of 6.4- to 6.8-day mouse embryos, the homeobox gene goosecoid is now shown to be expressed in a later phase of mouse development, from 10.5 days onwards. The later, spatially restricted domains of goosecoid expression are detected in the head, limbs and ventrolateral body wall. At all sites, the domains of expression are first detected in undifferentiated tissue, and then expression persists as these tissues undergo subsequent morphogenesis. For example, goosecoid expression is noted in the first branchial arch at 10.5 days, and then expression persists as this tissue undergoes morphogenesis to form the lower jaw and the body of the tongue. Expression in tissues around the first branchial cleft persists as these undergo morphogenesis to form the base of the auditory meatus and eustachian tube. Expression in tissues around the newly formed nasal pits persists as these elongate to form the nasal chambers. Expression in the ventral epithelial lining of the otic vesicle persists as this eventually gives rise to the non-sensory epithelium of the cochlea. Expression in the proximal limb buds and ventrolateral body wall persists as these tissues undergo morphogenesis to form proximal limb structures and ventral ribs respectively. Our findings lead us to suggest that the goosecoid gene product plays a role in spatial programming within discrete embryonic fields, and possibly lineage compartments, during organogenesis stages of mouse development.


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