The Effect of Diet on Craniofacial Growth in Osteogenesis Imperfecta Mouse Model

dc.contributor.advisorMenegaz, Rachel A.
dc.contributor.committeeMemberMaddux, Scott D.
dc.contributor.committeeMemberReeves, Rustin E.
dc.contributor.committeeMemberBorejdo, Julian
dc.creatorLadd, Summer H.
dc.date.accessioned2019-09-30T15:17:57Z
dc.date.available2019-09-30T15:17:57Z
dc.date.issued2018-05
dc.description.abstractOsteogenesis imperfecta (OI, or "brittle bone disease") is a rare disorder that is caused by genetic point mutations (COL1A1/COL1A2) that affect type 1 collagen. In OI type III (severe) patients, limb bones are more susceptible to skeletal fractures and the bones of the craniofacial region are underdeveloped. Some OI type III patients also suffer from dental malocclusions or fractures (dentinogenesis imperfecta, DI). The goals of this project are 1) to describe the facial phenotype in an OI mouse model, to see if this model can be used to test potential behavioral and pharmaceutical interventions; and 2) to determine if diet and masticatory loading affect the development of the craniofacial region in the OI model. The homozygous OI murine (OIM-/-), a mouse strain with a nonlethal recessively inherited mutation of the COL1A2 gene, is a potential model for the human OI type III. OIM-/- and wild type (WT) littermates were raised from weaning (21 days) to adulthood (16 weeks). Digital 3D craniofacial landmarks were taken from in-vivo micro CT scans, and Kuskal-Wallis ANOVAs, along with Mann-Whitney tests, were used to compare centroid size and interlandmark distances among treatment groups. This practicum focuses on the Week 10 mice, with 3 treatment groups: OIMxM, WTxM, and WTxP. We acknowledge that the sample is incomplete due to factors beyond our immediate control, such as OIM-/- survivability. Adolescent OIM-/- mice (week 10) were found to have on average smaller cranial and mandibular centroid sizes compared to WT mice regardless of diet. Week 10 OIM-/- mice also show several morphological similarities to the OI type III human phenotype, such as shortened cranial vault height, shortened jaw length, and altered dental spacing secondary to a shortened tooth row. We conclude that the OIM mouse model shows potential for future investigations of the growth mechanisms underlying the craniofacial presentation of OI. Furthermore, preliminary results suggest that masticatory loading during the early growth period can be used to stimulate craniofacial bone growth and improve bone quality in the OIM mouse model. Future studies will continue to improve sample size by treatment and age groups. The significance of this project is that it will give a better understanding of the role of type 1 collagen and the biomechanical mechanics of craniofacial development, which are important in the search for a new treatment method in OI.
dc.format.mimetypeapplication/pdf
dc.identifier.urihttps://hdl.handle.net/20.500.12503/29687
dc.language.isoen
dc.subjectbone
dc.subjectcollagen
dc.subjectcraniofacial phenotype
dc.subjectfunctional adaptation
dc.subjectosteogenesis imperfecta
dc.subjectskeletal growth
dc.subject.meshOsteogenesis Imperfecta
dc.subject.meshBone Development
dc.subject.meshBone and Bones
dc.subject.meshCollagen
dc.titleThe Effect of Diet on Craniofacial Growth in Osteogenesis Imperfecta Mouse Model
dc.typeThesis
dc.type.materialtext
thesis.degree.departmentGraduate School of Biomedical Sciences
thesis.degree.disciplineBiomedical Sciences
thesis.degree.grantorUniversity of North Texas Health Science Center at Fort Worth
thesis.degree.nameMaster of Science

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