Cranio-facial dysmorphism: experimental study in the mouse, clinical applications.

Abstract

To obtain a better understanding of mandibulo-facial dysostosis and hemicraniofacial microsomia in man, the authors carried out a histologic and scanning electron microscope study of the facial malformations produced in mouse embryos by retinoic acid and methyl-triazene. The administration of 400 mg/kg 13 cis-retinoic acid (RA) to pregnant C57BL mice on day 9 of gestation produced anomalies of the cephalic extremity in the embryos resembling human mandibulo-facial dysostosis. The 64 embryos collected presented hypoplasia of the branchial arches or the snout in 79% of cases, auricular anomalies in 47% and ophthalmic anomalies in 12.5%. Fourteen NMRI mice on day 10.5 of gestation were treated with 1.5 mg (0.5 mg/kg) methyl-triazene (Methyl). The 126 embryos collected had developed a very high percentage of micromandibles and anomalies of both embryonic ears (94.6% to 100%). Finally, although the facial anomalies produced by retinoic acid resemble the human mandibulo-facial dysostosis syndrome, no correlation was found between hemicraniofacial microsomia and the administration of methyl-triazene.

• Authors: R Glineur, S Louryan, A Lemaître, L Evrard, M Rooze, L De Vos
• Journal: Surgical and radiologic anatomy : SRA (Surg Radiol Anat) Vol. 21 Issue 1 Pg. 41-7 ( 1999) ISSN: 0930-1038 [Print] Germany
• PMID: 10370992 (Publication Type: Journal Article)

Chemical References

• Triazenes
• Tretinoin

Topics

• Abnormalities, Drug-Induced (embryology)
• Adolescent
• Animals
• Child
• Facial Asymmetry (chemically induced, embryology)
• Female
• Humans
• Male
• Mandibulofacial Dysostosis (chemically induced, embryology)
• Mice
• Mice, Inbred C57BL
• Microscopy, Electron, Scanning
• Pregnancy
• Tretinoin
• Triazenes