subject:(Shroom3)

McMaster University

1. KITALA, PATRICIA. Characterization of the Role of Shroom3 in Nephron Formation.

Degree: MSMS, 2019, McMaster University

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Proper development of the nephron, the functional unit of the kidney, is essential for kidney function. The nephron develops from a pool of cap mesenchymal… (more)

Subjects/Keywords: Shroom3; Kidney

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APA (6^th Edition):

KITALA, P. (2019). Characterization of the Role of Shroom3 in Nephron Formation. (Masters Thesis). McMaster University. Retrieved from http://hdl.handle.net/11375/24826

Chicago Manual of Style (16^th Edition):

KITALA, PATRICIA. “Characterization of the Role of Shroom3 in Nephron Formation.” 2019. Masters Thesis, McMaster University. Accessed February 27, 2021. http://hdl.handle.net/11375/24826.

MLA Handbook (7^th Edition):

KITALA, PATRICIA. “Characterization of the Role of Shroom3 in Nephron Formation.” 2019. Web. 27 Feb 2021.

Vancouver:

KITALA P. Characterization of the Role of Shroom3 in Nephron Formation. [Internet] [Masters thesis]. McMaster University; 2019. [cited 2021 Feb 27]. Available from: http://hdl.handle.net/11375/24826.

Council of Science Editors:

KITALA P. Characterization of the Role of Shroom3 in Nephron Formation. [Masters Thesis]. McMaster University; 2019. Available from: http://hdl.handle.net/11375/24826

McMaster University

2. Khalili, Hadiseh. SHROOM3 IN THE KIDNEY.

Degree: MSc, 2015, McMaster University

URL: http://hdl.handle.net/11375/17413

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Chronic kidney disease (CKD), defined as an irreversible reduction in glomerular filtration rate, is a large public health concern. Dissecting the genetic components of CKD… (more)

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Chronic kidney disease (CKD), defined as an irreversible reduction in glomerular filtration rate, is a large public health concern. Dissecting the genetic components of CKD is required to improve our understanding of disease pathogenesis. Researchers have identified that SHROOM3, has very high associations with kidney disease and function. Shroom3 encodes an actin-binding protein important in regulating cell and tissue morphogenesis. However, there is a lack of evidence supporting a role for Shroom3 in kidney function or disease. Here, I investigated the developmental and functional role of Shroom3 in the mammalian kidney. For the first time, I described the expression pattern of Shroom3 in the embryonic and adult mouse kidneys. By performing in situ hybridization and immunohistochemistry, I demonstrated that Shroom3 is expressed in the condensing mesenchyme, podocytes, and collecting ducts. I further showed that Shroom3 protein is localized in the foot processes of podocytes, utilizing immunogold labeling and transmission electron microscopy. In order to uncover a potential role of Shroom3 in the kidney, we utilized Shroom3 knockout mice. Shroom3 mutants demonstrated marked glomerular abnormalities including cystic and degenerating glomeruli, and reduced glomerular number. Scanning and transmission electron microscopic analyses of Shroom3 mutant glomeruli revealed disruptions in podocyte morphology characterized by disorganized foot processes with less interdigitation and segmental foot processes effacement. Furthermore, immunofluorescence analysis of mutant kidneys revealed aberrant distribution of podocyte actin-associated proteins. Elucidating the underlying molecular mechanism of this abnormal podocyte architecture; v we demonstrated that in the absence of Shroom3, Rho kinase is mislocalized in the apical membrane of podocytes. As a result, mislocalized Rho kinase failed to phosphorylate non-muscle myosin and induce actomyosin contraction resulting in a patchy granular distribution of actin in the podocytes of Shroom3 mutants. Taken together, our findings established that Shroom3 is essential for proper actin organization in the podocytes through interaction with Rock. Furthermore, we took advantage of a haploinsufficiency phenotype of Shroom3 heterozygote adult mice and demonstrated these mice develop glomerulosclerosis and proteinuria. In conclusion, our studies provided evidence to support a role for Shroom3 in kidney development and disease and support the GWAS studies that suggested a correlation between SHROOM3 variants and kidney function in humans.

Thesis

Master of Science (MSc)

Advisors/Committee Members: Bridgewater, Darren, Medical Sciences.

Subjects/Keywords: Kidney; Glomerulus; Podocyte; Shroom3; CKD; Kidney Development

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APA (6^th Edition):

Khalili, H. (2015). SHROOM3 IN THE KIDNEY. (Masters Thesis). McMaster University. Retrieved from http://hdl.handle.net/11375/17413

Chicago Manual of Style (16^th Edition):

Khalili, Hadiseh. “SHROOM3 IN THE KIDNEY.” 2015. Masters Thesis, McMaster University. Accessed February 27, 2021. http://hdl.handle.net/11375/17413.

MLA Handbook (7^th Edition):

Khalili, Hadiseh. “SHROOM3 IN THE KIDNEY.” 2015. Web. 27 Feb 2021.

Vancouver:

Khalili H. SHROOM3 IN THE KIDNEY. [Internet] [Masters thesis]. McMaster University; 2015. [cited 2021 Feb 27]. Available from: http://hdl.handle.net/11375/17413.

Council of Science Editors:

Khalili H. SHROOM3 IN THE KIDNEY. [Masters Thesis]. McMaster University; 2015. Available from: http://hdl.handle.net/11375/17413

University of Western Ontario

3. Sull, Alexandra. The Role of Shroom3 in Kidney Development.

Degree: 2015, University of Western Ontario

URL: https://ir.lib.uwo.ca/etd/2881

► Chronic kidney disease (CKD) affects 10% of the population in industrialized nations. Recent genome wide association studies (GWAS) have correlated SHROOM3 with glomerular filtration rate… (more)

Subjects/Keywords: Shroom3; nephrogenesis; chronic kidney disease; actin; podocyte; mouse; Other Physiology

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APA (6^th Edition):

Sull, A. (2015). The Role of Shroom3 in Kidney Development. (Thesis). University of Western Ontario. Retrieved from https://ir.lib.uwo.ca/etd/2881

Note: this citation may be lacking information needed for this citation format:
Not specified: Masters Thesis or Doctoral Dissertation

Chicago Manual of Style (16^th Edition):

Sull, Alexandra. “The Role of Shroom3 in Kidney Development.” 2015. Thesis, University of Western Ontario. Accessed February 27, 2021. https://ir.lib.uwo.ca/etd/2881.

Note: this citation may be lacking information needed for this citation format:
Not specified: Masters Thesis or Doctoral Dissertation

MLA Handbook (7^th Edition):

Sull, Alexandra. “The Role of Shroom3 in Kidney Development.” 2015. Web. 27 Feb 2021.

Vancouver:

Sull A. The Role of Shroom3 in Kidney Development. [Internet] [Thesis]. University of Western Ontario; 2015. [cited 2021 Feb 27]. Available from: https://ir.lib.uwo.ca/etd/2881.

Note: this citation may be lacking information needed for this citation format:
Not specified: Masters Thesis or Doctoral Dissertation

Council of Science Editors:

Sull A. The Role of Shroom3 in Kidney Development. [Thesis]. University of Western Ontario; 2015. Available from: https://ir.lib.uwo.ca/etd/2881

Note: this citation may be lacking information needed for this citation format:
Not specified: Masters Thesis or Doctoral Dissertation

University of Western Ontario

4. Ko, Doyun. Functional Changes Associated with SHROOM3 Deficiency and Mutation: Implications in Renal Pathophysiology and Disease.

Degree: 2018, University of Western Ontario

URL: https://ir.lib.uwo.ca/etd/5245

► Chronic kidney disease (CKD) is a substantial global health burden that has recently been linked to the genetic locus SHROOM3 by several genome-wide association studies.… (more)

Subjects/Keywords: Shroom3; kidney; chronic kidney disease; hypertension; salt; blood pressure; Cell and Developmental Biology; Diseases; Physiology

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APA (6^th Edition):

Ko, D. (2018). Functional Changes Associated with SHROOM3 Deficiency and Mutation: Implications in Renal Pathophysiology and Disease. (Thesis). University of Western Ontario. Retrieved from https://ir.lib.uwo.ca/etd/5245

Note: this citation may be lacking information needed for this citation format:
Not specified: Masters Thesis or Doctoral Dissertation

Chicago Manual of Style (16^th Edition):

Ko, Doyun. “Functional Changes Associated with SHROOM3 Deficiency and Mutation: Implications in Renal Pathophysiology and Disease.” 2018. Thesis, University of Western Ontario. Accessed February 27, 2021. https://ir.lib.uwo.ca/etd/5245.

Note: this citation may be lacking information needed for this citation format:
Not specified: Masters Thesis or Doctoral Dissertation

MLA Handbook (7^th Edition):

Ko, Doyun. “Functional Changes Associated with SHROOM3 Deficiency and Mutation: Implications in Renal Pathophysiology and Disease.” 2018. Web. 27 Feb 2021.

Vancouver:

Ko D. Functional Changes Associated with SHROOM3 Deficiency and Mutation: Implications in Renal Pathophysiology and Disease. [Internet] [Thesis]. University of Western Ontario; 2018. [cited 2021 Feb 27]. Available from: https://ir.lib.uwo.ca/etd/5245.

Note: this citation may be lacking information needed for this citation format:
Not specified: Masters Thesis or Doctoral Dissertation

Council of Science Editors:

Ko D. Functional Changes Associated with SHROOM3 Deficiency and Mutation: Implications in Renal Pathophysiology and Disease. [Thesis]. University of Western Ontario; 2018. Available from: https://ir.lib.uwo.ca/etd/5245

Note: this citation may be lacking information needed for this citation format:
Not specified: Masters Thesis or Doctoral Dissertation

The Ohio State University

5. Heimlich, Derek. Temporal Examination and Quantification of Fiber Cell Morphology and Arrangement in Chick and Mouse Lenses.

Degree: MS, Vision Science, 2020, The Ohio State University

URL: http://rave.ohiolink.edu/etdc/view?acc_num=osu1586982635479627

► The transparency and elasticity of the crystalline lens is a result of the configuration of regularly assembled hexagonal secondary lens fibers that are continuously added… (more)

Subjects/Keywords: Developmental Biology; Ophthalmology; Lens Fiber Morphology; Shroom3; p-120 catenin; rho-kinase; Lens development

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APA · Chicago · MLA · Vancouver · CSE | Export to Zotero / EndNote / Reference Manager

APA (6^th Edition):

Heimlich, D. (2020). Temporal Examination and Quantification of Fiber Cell Morphology and Arrangement in Chick and Mouse Lenses. (Masters Thesis). The Ohio State University. Retrieved from http://rave.ohiolink.edu/etdc/view?acc_num=osu1586982635479627

Chicago Manual of Style (16^th Edition):

Heimlich, Derek. “Temporal Examination and Quantification of Fiber Cell Morphology and Arrangement in Chick and Mouse Lenses.” 2020. Masters Thesis, The Ohio State University. Accessed February 27, 2021. http://rave.ohiolink.edu/etdc/view?acc_num=osu1586982635479627.

MLA Handbook (7^th Edition):

Heimlich, Derek. “Temporal Examination and Quantification of Fiber Cell Morphology and Arrangement in Chick and Mouse Lenses.” 2020. Web. 27 Feb 2021.

Vancouver:

Heimlich D. Temporal Examination and Quantification of Fiber Cell Morphology and Arrangement in Chick and Mouse Lenses. [Internet] [Masters thesis]. The Ohio State University; 2020. [cited 2021 Feb 27]. Available from: http://rave.ohiolink.edu/etdc/view?acc_num=osu1586982635479627.

Council of Science Editors:

Heimlich D. Temporal Examination and Quantification of Fiber Cell Morphology and Arrangement in Chick and Mouse Lenses. [Masters Thesis]. The Ohio State University; 2020. Available from: http://rave.ohiolink.edu/etdc/view?acc_num=osu1586982635479627

6. Chung, Mei-I. Transcriptional control of epithelial morphogenesis.

Degree: PhD, Cell and Molecular Biology, 2013, University of Texas – Austin

URL: http://hdl.handle.net/2152/25166

► How tissues and organs develop into their final shape during embryogenesis is a fascinating and long-standing question in developmental biology. Tissue morphogenesis is driven by… (more)

▼ How tissues and organs develop into their final shape during embryogenesis is a fascinating and long-standing question in developmental biology. Tissue morphogenesis is driven by a variety of events at the cellular level and individual cell shape change is one of the central morphogenetic engines. Thus, it is crucial to understand what signals specify the correct cell behavior in specific groups of cells during development. For my doctoral studies, I have focused on two cell shape change events, apical constriction and cilia assembly. First, we present data demonstrating that Shroom3 is essential for cell shape changes and morphogenesis in the developing vertebrate gut, where Shroom3 transcription requires the Pitx1 transcription factor. We identified a Pitx-responsive regulatory element in the genomic DNA upstream of Shroom3, and showed that Pitx proteins directly activated Shroom3 transcription in Xenopus. Moreover, we showed that ectopic expression of Pitx proteins was sufficient to induce Shroom3-dependent cytoskeletal reorganization and epithelial cell shape change. These data demonstrated new breadth to the requirements for Shroom3 in morphogenesis, and also provided a cell-biological mechanism for Pitx transcription factors action during morphogenesis. Next, we focused on understanding the transcriptional regulation of ciliogenesis. We first showed that Rfx2 transcription factor broadly controlled ciliogenesis, and by RNA- and ChIP-sequencing, we showed that Rfx2 directly regulated a wide range of genes encoding diverse ciliogenic machinery. Finally, in addition to ciliogenesis regulation, a large number of non-ciliary genes in our Rfx2 dataset led us to identify a novel role of Rfx2 in controlling insertion of multi-ciliated cells into the overlying mucociliary epithelium. Moreover, we showed here that Slit2, a target of Rfx2, was involved in multi-ciliated cell movements, possibly through mediating cortical E-cadherin level. This work allowed us to begin building a genetic network controlling multi-ciliated cells in mucociliary epithelium. Together, we showed a transcriptional regulation of apical constriction driving gut morphogenesis and a comprehensive transcriptional network that governs multi-ciliated cell development. Advisors/Committee Members: Wallingford, John B. (advisor).

Subjects/Keywords: Morphogenesis; Shroom3; Rfx2; Cilia; Transcription; Xenopus

…changes in cell behavior are Shroom3 dependent 29 2.2.9 Pitx1 does not activate the expression… …of genes encoding known Shroom3 effector proteins… …7 Figure 2.1. Shroom3 is essential for the cell shape change during the gut… …Figure 2.2. Pitx1 and Shroom3 are co-expressed in the developing gut...............20 Figure… …2.3. Pitx1controls Shroom3 expression in the developing gut ..................21 Figure 2.4…

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APA (6^th Edition):

Chung, M. (2013). Transcriptional control of epithelial morphogenesis. (Doctoral Dissertation). University of Texas – Austin. Retrieved from http://hdl.handle.net/2152/25166

Chicago Manual of Style (16^th Edition):

Chung, Mei-I. “Transcriptional control of epithelial morphogenesis.” 2013. Doctoral Dissertation, University of Texas – Austin. Accessed February 27, 2021. http://hdl.handle.net/2152/25166.

MLA Handbook (7^th Edition):

Chung, Mei-I. “Transcriptional control of epithelial morphogenesis.” 2013. Web. 27 Feb 2021.

Vancouver:

Chung M. Transcriptional control of epithelial morphogenesis. [Internet] [Doctoral dissertation]. University of Texas – Austin; 2013. [cited 2021 Feb 27]. Available from: http://hdl.handle.net/2152/25166.

Council of Science Editors:

Chung M. Transcriptional control of epithelial morphogenesis. [Doctoral Dissertation]. University of Texas – Austin; 2013. Available from: http://hdl.handle.net/2152/25166

7. Halabi, Rami R. Shroom3 deficient mice show congenital heart defects.

Degree: 2013, University of Western Ontario

URL: https://ir.lib.uwo.ca/etd/1394

► Congenital heart defects (CHDs) are associated with a number of genetic and environmental risk factors affecting approximately 1% of newborns. Shroom3 is an actin binding… (more)

Subjects/Keywords: mice; cardiogenesis; Shroom3; congenital heart defects; actin; cytoskeleton; Developmental Biology

…3.10 Shroom3 deficient embryos have reduced heart function… …71 4.1 Shroom3 is expressed early in heart development… …71 4.2 Shroom3 is required for normal cardiac development… …73 4.3 Shroom3 alters cellular but not cytoskeletal morphology… …77 4.4 Shroom3 is required for normal cardiac function…

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APA (6^th Edition):

Halabi, R. R. (2013). Shroom3 deficient mice show congenital heart defects. (Thesis). University of Western Ontario. Retrieved from https://ir.lib.uwo.ca/etd/1394

Note: this citation may be lacking information needed for this citation format:
Not specified: Masters Thesis or Doctoral Dissertation

Chicago Manual of Style (16^th Edition):

Halabi, Rami R. “Shroom3 deficient mice show congenital heart defects.” 2013. Thesis, University of Western Ontario. Accessed February 27, 2021. https://ir.lib.uwo.ca/etd/1394.

Note: this citation may be lacking information needed for this citation format:
Not specified: Masters Thesis or Doctoral Dissertation

MLA Handbook (7^th Edition):

Halabi, Rami R. “Shroom3 deficient mice show congenital heart defects.” 2013. Web. 27 Feb 2021.

Vancouver:

Halabi RR. Shroom3 deficient mice show congenital heart defects. [Internet] [Thesis]. University of Western Ontario; 2013. [cited 2021 Feb 27]. Available from: https://ir.lib.uwo.ca/etd/1394.

Note: this citation may be lacking information needed for this citation format:
Not specified: Masters Thesis or Doctoral Dissertation

Council of Science Editors:

Halabi RR. Shroom3 deficient mice show congenital heart defects. [Thesis]. University of Western Ontario; 2013. Available from: https://ir.lib.uwo.ca/etd/1394

Note: this citation may be lacking information needed for this citation format:
Not specified: Masters Thesis or Doctoral Dissertation

8. Eckes, Melissa. Shroom3 Localization and Apical Constriction during the Development of the Crystalline Lens in Mouse Embryos.

Degree: MS, Vision Science, 2017, The Ohio State University

URL: http://rave.ohiolink.edu/etdc/view?acc_num=osu1492728223255337

► Purpose:The purpose of this research was to analyze the localization of the protein Shroom3, and determine its role in regulating lens fiber cell shape during… (more)

Subjects/Keywords: Developmental Biology; Shroom3 protein; Crystalline Lens development; mouse embryos

…3 Apical Constriction and Shroom3… …23 Shroom3 Localization with X-gal Staining… …23 Shroom3 Localization with Antibody Staining… …34 Localization of Shroom3… …Shroom3 null embryos 29 Table 2. Ratio of average apical width to average basal width in…

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APA (6^th Edition):

Eckes, M. (2017). Shroom3 Localization and Apical Constriction during the Development of the Crystalline Lens in Mouse Embryos. (Masters Thesis). The Ohio State University. Retrieved from http://rave.ohiolink.edu/etdc/view?acc_num=osu1492728223255337

Chicago Manual of Style (16^th Edition):

Eckes, Melissa. “Shroom3 Localization and Apical Constriction during the Development of the Crystalline Lens in Mouse Embryos.” 2017. Masters Thesis, The Ohio State University. Accessed February 27, 2021. http://rave.ohiolink.edu/etdc/view?acc_num=osu1492728223255337.

MLA Handbook (7^th Edition):

Eckes, Melissa. “Shroom3 Localization and Apical Constriction during the Development of the Crystalline Lens in Mouse Embryos.” 2017. Web. 27 Feb 2021.

Vancouver:

Eckes M. Shroom3 Localization and Apical Constriction during the Development of the Crystalline Lens in Mouse Embryos. [Internet] [Masters thesis]. The Ohio State University; 2017. [cited 2021 Feb 27]. Available from: http://rave.ohiolink.edu/etdc/view?acc_num=osu1492728223255337.

Council of Science Editors:

Eckes M. Shroom3 Localization and Apical Constriction during the Development of the Crystalline Lens in Mouse Embryos. [Masters Thesis]. The Ohio State University; 2017. Available from: http://rave.ohiolink.edu/etdc/view?acc_num=osu1492728223255337

9. Lee, Chan-jae. The roles of Shroom family proteins during Xenopus development.

Degree: PhD, Cell and Molecular Biology, 2009, University of Texas – Austin

URL: http://hdl.handle.net/2152/6544

► The Shroom family of proteins is currently comprised of four members, Shroom1, 2, 3 and 4. Since Shroom3 was shown to be a critical protein… (more)

▼ The Shroom family of proteins is currently comprised of four members, Shroom1, 2, 3 and 4. Since Shroom3 was shown to be a critical protein for neural tube closure, the other three proteins are also expected to play an important role for proper development. However, their functions during development were not clear. To address this, my study started with Shroom3 function in the neural plate. Shroom3 had been previously known to induce apical constriction by controlling actin filaments in neuroepithelial cells. My studies show that Shroom3 induces apico-basal cell heightening by controlling parallel microtubule assembly. Shroom3 is able to change the distribution of γ-tubulin, suggesting that Shroom3 controls apical constriction and apico-basal cell elongation via both actin filaments and microtubules. The ability to control γ-tubulin distribution is possessed not only by Shroom3, but also by all other Shroom proteins, although they can not induce apical constriction. In addition, they are expressed in tissues which contain apico-basally elongated cells. Data from functional assays with Shroom2 show that it induces cell elongation and is required for proper cell shape in deep layer neuroepithelial cells in Xenopus. These data suggest that Shroom family proteins control cell architecture during morphogenetic development. I have discovered another role for Shroom2. By comparative analysis with Xenopus and Physalaemus, which have different pigment patterns in eggs, I show that a high level of maternal Shroom2 mRNA is important for pigment polarity in Xenopus. Furthermore, Shroom2 controls the distribution of spectrin which plays a role in pigment granule movement. Thus, Shroom2 is suggested to be a key molecule to control the pigment polarity in amphibian eggs. Together all these data suggest that Shroom family proteins play a role in cell morphogenesis and polarization via controlling the cytoskeleton during Xenopus development. Advisors/Committee Members: Wallingford, John B. (advisor).

Subjects/Keywords: Shroom family proteins; Xenopus development; Shroom3; Shroom2; Cell morphogenesis; Polarization; Pigment polarity

…II. 2. 2. Shroom3 controls apical constriction in all regions of Xenopus neural plate… …16 II. 2. 3. Shroom3 is required for cell elongation in the neural plate of Xenopus… …neuroepithelial cells ...................................................24 II. 2. 5. Shroom3 controls… …the distribution of γ-tubulin ......................27 II. 2. 6. Ectopic Shroom3 expression… …31 II. 2. 7. Shroom3 controls megalin distribution .................................33 II…

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APA · Chicago · MLA · Vancouver · CSE | Export to Zotero / EndNote / Reference Manager

APA (6^th Edition):

Lee, C. (2009). The roles of Shroom family proteins during Xenopus development. (Doctoral Dissertation). University of Texas – Austin. Retrieved from http://hdl.handle.net/2152/6544

Chicago Manual of Style (16^th Edition):

Lee, Chan-jae. “The roles of Shroom family proteins during Xenopus development.” 2009. Doctoral Dissertation, University of Texas – Austin. Accessed February 27, 2021. http://hdl.handle.net/2152/6544.

MLA Handbook (7^th Edition):

Lee, Chan-jae. “The roles of Shroom family proteins during Xenopus development.” 2009. Web. 27 Feb 2021.

Vancouver:

Lee C. The roles of Shroom family proteins during Xenopus development. [Internet] [Doctoral dissertation]. University of Texas – Austin; 2009. [cited 2021 Feb 27]. Available from: http://hdl.handle.net/2152/6544.

Council of Science Editors:

Lee C. The roles of Shroom family proteins during Xenopus development. [Doctoral Dissertation]. University of Texas – Austin; 2009. Available from: http://hdl.handle.net/2152/6544

10. Lappin, Cory James. Investigating the Role of Shroom3 in Collagen Regulation and Development of the Corneal Stroma.

Degree: MS, Vision Science, 2018, The Ohio State University

URL: http://rave.ohiolink.edu/etdc/view?acc_num=osu1523921114811659

► Purpose:The cornea stroma consists of collagen fibrils that exist in a highly ordered structure, however the mechanisms by which collagen expression and its complex… (more)

▼ Purpose:The cornea stroma consists of collagen fibrils that exist in a highly ordered structure, however the mechanisms by which collagen expression and its complex architecture are regulated are not well understood. Insight into the mechanisms that underlie collagen expression and fibril architecture would shed light onto the etiologies of corneal diseases. Thus, identifying specific pathways or proteins that are in involved in stromal collagen regulation could help better explain how collagen is regulated in health, and what goes wrong in cases of pathology. A protein recently shown to induce collagen expression is the cytoplasmic protein called Shroom3. This F-actin and Rho-kinase binding protein is best known for regulating epithelial cell shape during embryonic morphogenesis, but the mechanisms by which it regulates collagen expression have not been widely studied. Previous research has suggested that abnormal SHROOM3 disrupts cornea development in mouse embryos and that a homozygous missense mutation in this gene has been associated with the stromal thinning disease known as keratoconus. Therefore, the purpose of this research was to further characterize the corneal phenotype in Shroom3-deficient embryos and discern what effect the keratoconus-associated mutation has on Shroom3 function. Methods:Embryonic corneal Shroom3 expression was observed using X-gal labeling of Shroom3+/- embryos that express X-galactosidase under the control of the endogenous Shroom3 promoter. To determine the function of Shroom3 during cornea development, control and Shroom3-deficient mouse embryos were analyzed using histological staining, immunofluorescent imaging, and electron microscopy to ascertain the effect on collagen expression, fibril size, keratocyte number, stromal thickness and corneal epithelial shape. The consequences of mutations on Shroom3 function were analyzed in kidney- (MDCK) and cornea-derived (SIRC) cells following transfection with plasmids expressing wild-type Shroom3 (Shroom3wt), a Rho-kinase binding deficient mutation (Shroom3R1838C) and the keratoconus-associated mutation (Shroom3G59V). Epithelial cell shape and collagen expression were analyzed by immunofluorescent imaging and RT-PCR analysis, and electron microscopy was performed to analyze the effect Shroom3 has on collagen expression, fibril size, and corneal phenotypes. Results:Shroom3 expression in the cornea was observed in the central and peripheral cornea, however expression was higher in the periphery and posterior stroma. Shroom3-/- cornea epithelial cells had larger apical areas compared to Shroom3+/- cells, suggesting reduced apical constriction. Shroom3-/- corneas displayed a reduction in total number of keratocytes in the central cornea. There was no difference in stromal thickness between Shroom3+/- and Shroom3-/- corneas when measured directly, but there was a reduction in the layers of keratocytes observed in Shroom3-/- corneas which acted as an indirect measure of stromal thickness. The intensity of… Advisors/Committee Members: Plageman, Timothy (Advisor).

Subjects/Keywords: Developmental Biology; Genetics; Ophthalmology; shroom3; shroom3 protein; cornea; stroma; corneal stroma; collagen; stromal collagen; keratocyte; keratoconus; PDZ; ASD2; G59V; R1838C; G60V; collagen fibril; epithelium; corneal epithelium; COL1; COL4; Col1a1; Col4a1; apical constriction; Rho-kinase

…10 Shroom3… …32 Shroom3 Localization… …43 Shroom3 Localization and Apical Constriction in Corneal Epithelial Cells… …43 Shroom3 and Peters’ Anomaly… …44 Shroom3 and Stromal Thickness…

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APA (6^th Edition):

Lappin, C. J. (2018). Investigating the Role of Shroom3 in Collagen Regulation and Development of the Corneal Stroma. (Masters Thesis). The Ohio State University. Retrieved from http://rave.ohiolink.edu/etdc/view?acc_num=osu1523921114811659

Chicago Manual of Style (16^th Edition):

Lappin, Cory James. “Investigating the Role of Shroom3 in Collagen Regulation and Development of the Corneal Stroma.” 2018. Masters Thesis, The Ohio State University. Accessed February 27, 2021. http://rave.ohiolink.edu/etdc/view?acc_num=osu1523921114811659.

MLA Handbook (7^th Edition):

Lappin, Cory James. “Investigating the Role of Shroom3 in Collagen Regulation and Development of the Corneal Stroma.” 2018. Web. 27 Feb 2021.

Vancouver:

Lappin CJ. Investigating the Role of Shroom3 in Collagen Regulation and Development of the Corneal Stroma. [Internet] [Masters thesis]. The Ohio State University; 2018. [cited 2021 Feb 27]. Available from: http://rave.ohiolink.edu/etdc/view?acc_num=osu1523921114811659.

Council of Science Editors:

Lappin CJ. Investigating the Role of Shroom3 in Collagen Regulation and Development of the Corneal Stroma. [Masters Thesis]. The Ohio State University; 2018. Available from: http://rave.ohiolink.edu/etdc/view?acc_num=osu1523921114811659

11. Lemay, Philippe. Anomalies du tube neural : mieux comprendre les causes génétiques de cette pathologie complexe.

Degree: 2018, Université de Montréal

URL: http://hdl.handle.net/1866/20746

Subjects/Keywords: Anomalies du tube neural; GRHL3; SHROOM3; Séquençage de l’exome entier; Génétique complexe; Mutations de novo; Neural tube defects; Whole exome sequencing; Complex genetics; De novo mutations; Biology - Genetics / Biologie - Génétique (UMI : 0369)

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APA (6^th Edition):

Lemay, P. (2018). Anomalies du tube neural : mieux comprendre les causes génétiques de cette pathologie complexe. (Thesis). Université de Montréal. Retrieved from http://hdl.handle.net/1866/20746

Note: this citation may be lacking information needed for this citation format:
Not specified: Masters Thesis or Doctoral Dissertation

Chicago Manual of Style (16^th Edition):

Lemay, Philippe. “Anomalies du tube neural : mieux comprendre les causes génétiques de cette pathologie complexe.” 2018. Thesis, Université de Montréal. Accessed February 27, 2021. http://hdl.handle.net/1866/20746.

Note: this citation may be lacking information needed for this citation format:
Not specified: Masters Thesis or Doctoral Dissertation

MLA Handbook (7^th Edition):

Lemay, Philippe. “Anomalies du tube neural : mieux comprendre les causes génétiques de cette pathologie complexe.” 2018. Web. 27 Feb 2021.

Vancouver:

Lemay P. Anomalies du tube neural : mieux comprendre les causes génétiques de cette pathologie complexe. [Internet] [Thesis]. Université de Montréal; 2018. [cited 2021 Feb 27]. Available from: http://hdl.handle.net/1866/20746.

Note: this citation may be lacking information needed for this citation format:
Not specified: Masters Thesis or Doctoral Dissertation

Council of Science Editors:

Lemay P. Anomalies du tube neural : mieux comprendre les causes génétiques de cette pathologie complexe. [Thesis]. Université de Montréal; 2018. Available from: http://hdl.handle.net/1866/20746

Note: this citation may be lacking information needed for this citation format:
Not specified: Masters Thesis or Doctoral Dissertation

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Open Access Theses and Dissertations