Cranio-facial dysmorphism: experimental study in the mouse, clinical applications.
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.
Glineur
R
R
Service de Chirurgie Maxillo-Faciale, Cliniques Universitaires, Hôpital Erasme, Brussels, Belgium.
Louryan
S
S
Lemaître
A
A
Evrard
L
L
Rooze
M
M
De Vos
L
L
D000014:Abnormalities, Drug-Induced / Q000196:embryology; D000293:Adolescent; D000818:Animals; D002648:Child; D005146:Facial Asymmetry / Q000139:chemically induced / Q000196:embryology*; D005260:Female; D006801:Humans; D008297:Male; D008342:Mandibulofacial Dysostosis / Q000139:chemically induced / Q000196:embryology*; D051379:Mice; D008810:Mice, Inbred C57BL; D008855:Microscopy, Electron, Scanning; D011247:Pregnancy; D014212:Tretinoin; D014226:Triazenes
D014226:Triazenes; D014212:Tretinoin