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Finding the Unicorn, a New Mouse Model of Midfacial Clefting

Lantz, Brandi and White, Casey and Liu, Xinyun and Wan, Yong and Gabriel, George and Lo, Cecilia W. Y. and Szabo-Rogers, Heather L. (2020) Finding the Unicorn, a New Mouse Model of Midfacial Clefting. Genes, 11 (1). p. 83. ISSN 2073-4425

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Human midfacial clefting is a rare subset of orofacial clefting and in severe cases, the cleft separates the nostrils splitting the nose into two independent structures. To begin to understand the morphological and genetic causes of midfacial clefting we recovered the Unicorn mouse line. Unicorn embryos develop a complete midfacial cleft through the lip, and snout closely modelling human midfacial clefting. The Unicorn mouse line has ethylnitrosourea (ENU)-induced missense mutations in Raldh2 and Leo1. The mutations segregate with the cleft face phenotype. Importantly, the nasal cartilages and surrounding bones are patterned and develop normal morphology, except for the lateral displacement because of the cleft. We conclude that the midfacial cleft arises from the failure of the medial convergence of the paired medial nasal prominences between E10.5 to E11.5 rather than defective cell proliferation and death. Our work uncovers a novel mouse model and mechanism for the etiology of midfacial clefting.


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Item Type: Article
Status: Published
CreatorsEmailPitt UsernameORCID
Lantz, Brandi
White, Casey
Liu, Xinyun
Wan, Yongyow11@pitt.eduyow11
Gabriel, Georgegcg9@pitt.edugcg90000-0003-3250-2518
Lo, Cecilia W. Y.cel36@pitt.educel360000-0003-4314-3434
Szabo-Rogers, Heather L.hsrogers@pitt.eduhsrogers0000-0002-7921-8031
Centers: Other Centers, Institutes, Offices, or Units > McGowan Institute for Regenerative Medicine
Date: 11 January 2020
Date Type: Publication
Journal or Publication Title: Genes
Volume: 11
Number: 1
Publisher: MDPI AG
Page Range: p. 83
DOI or Unique Handle: 10.3390/genes11010083
Schools and Programs: School of Dental Medicine > Dental Science
Refereed: Yes
Uncontrolled Keywords: midfacial clefting, unicorn, medial nasal prominences, nasal septum, raldh2, leo1
ISSN: 2073-4425
Official URL:
Funders: University of Pittsburgh School of Dental Medicine
Article Type: Research Article
Date Deposited: 05 Jan 2022 16:40
Last Modified: 05 Jan 2022 16:40


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