Date on Master's Thesis/Doctoral Dissertation
5-2019
Document Type
Master's Thesis
Degree Name
M.S.
Department
Oral Biology
Degree Program
Oral Biology, MS
Committee Chair
Sandell, Lisa
Committee Co-Chair (if applicable)
Wang, Huizhi
Committee Member
Wang, Huizhi
Committee Member
Ding, Jixiang
Committee Member
Bagaitkar, Juhi
Author's Keywords
retinoic acid; cleft palate; spontaneous mouth movement; RDH10; vitamin A
Abstract
Cleft palate is a common birth defect. Etiologic mechanisms of palate cleft include defects in palate morphogenesis, mandibular growth, or spontaneous fetal mouth movement. Cleft palate linked to deficient fetal mouth movement has been demonstrated directly only in a single experimental model of loss of neurotransmission. Here, using retinoid deficient mouse embryos, we demonstrate directly for the first time that deficient fetal mouth movement and cleft palate occurs as a result of mis-patterned development of pharyngeal peripheral nerves and cartilages. Retinoid deficient embryos were generated by inactivation of retinol dehydrogenase 10 (Rdh10), which is critical for production of Retinoic Acid (RA) during embryogenesis. Using X-ray microtomography (microCT), in utero ultrasound, ex vivo culture, and tissue staining, we demonstrate that retinoid deficient mouse embryos lack fetal mouth movement owing to mis-patterning of pharyngeal cartilages and motor nerves. Findings from this study may indicate the earliest marker for diagnosing cleft palate.
Recommended Citation
Friedl, Regina, "Defects in fetal mouth movement and pharyngeal patterning underlie cleft palate caused by retinoid deficiency." (2019). Electronic Theses and Dissertations. Paper 3157.
https://doi.org/10.18297/etd/3157
Included in
Animal Structures Commons, Biology Commons, Developmental Biology Commons, Developmental Neuroscience Commons, Disease Modeling Commons, Embryonic Structures Commons, Nervous System Commons
