Lee, Eunsol
(2020)
RELATIONSHIP BETWEEN CRANIAL BASE DEVELOPMENT AND CLEFT LIP AND PALATE IN PRICKLE1 BEETLEJUICE MUTANT: CASE CONTROL STUDY.
Master's Thesis, University of Pittsburgh.
(Unpublished)
Abstract
Cleft lip and/or palate (CL/P) is one of the most common congenital anomalies in United States. Its etiology is complex, multifactorial, and not well understood. This study focuses on Prickle1 Beetlejuice (BJ) mutants which tend to have compressed and wide facial morphology. Prickle1 is a core component of Wnt/Planar cell polarity(PCP) pathway and the Prickle1Bj mouse line has a missense mutation (p.Cys161Phe) that disrupts the LIM1 domain in Prickle1. These mutants have approximately 50% chance of developing a cleft palate. Because cranial base size and shape determine the perimeter of growth for the lower 2/3 of the face, we examine the association between cranial base development and orofacial cleft. We found that Prickle1Bj/Bj with cleft lip and/or palate have wider, shorter, and less dense basisphenoid compared to wild type. Mutants with both cleft lip and palate compared to mutants with cleft lip only have even shorter and less dense basisphenoid. However, the basisphenoid width difference between the mutant groups was not statistically significant. Our data supports the conclusion that wide basal cranium poses higher risk of developing orofacial cleft. Yet, basisphenoid bone density is the superior value in determining the degree of orofacial cleft.
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Details
| Item Type: |
University of Pittsburgh ETD
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| Status: |
Unpublished |
| Creators/Authors: |
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| ETD Committee: |
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| Date: |
13 July 2020 |
| Date Type: |
Publication |
| Defense Date: |
13 February 2020 |
| Approval Date: |
13 July 2020 |
| Submission Date: |
19 May 2020 |
| Access Restriction: |
No restriction; Release the ETD for access worldwide immediately. |
| Number of Pages: |
45 |
| Institution: |
University of Pittsburgh |
| Schools and Programs: |
School of Dental Medicine > Dental Science |
| Degree: |
MDS - Master of Dental Science |
| Thesis Type: |
Master's Thesis |
| Refereed: |
Yes |
| Uncontrolled Keywords: |
Cleft lip, cleft palate, orofacial cleft, Prickle1, Wnt, Planar cell polarity, PCP, cranial base development, basisphenoid |
| Date Deposited: |
13 Jul 2020 18:01 |
| Last Modified: |
13 Jul 2020 18:01 |
| URI: |
http://d-scholarship.pitt.edu/id/eprint/39061 |
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