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Journal of Dental Research, Vol. 82, No. 12, 1013-1017 (2003)
DOI: 10.1177/154405910308201215

MSX1 Gene is Deleted in Wolf-Hirschhorn Syndrome Patients with Oligodontia

P. Nieminen1,5,*, J. Kotilainen1,3, Y. Aalto4, S. Knuutila4, S. Pirinen1,2 and I. Thesleff5,3

1 Institute of Dentistry, Biomedicum, PO Box 63, FIN-00014 University of Helsinki, Finland;
2 Department of Oral and Maxillofacial Diseases, Helsinki University Central Hospital, Finland;
3 Department of Health, City of Helsinki, Finland;
4 Department of Medical Genetics, Haartman Institute, Helsinki University Central Hospital, University of Helsinki, Finland; and
5 Institute of Biotechnology, University of Helsinki, Finland;


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  		Figure. Tooth phenotype in Wolf-Hirschhorn syndrome. (A) Panoramic tomogram of a 8.7-year-old Wolf-Hirschhorn female (patient #4) with only four molars, lower first premolars, upper left canine and central incisors, and four lower incisors of the permanent dentition developing (arrows). Presence of upper right lateral incisor and canine was confirmed with periapical x-ray. Molars were judged to be second molars and four teeth in the anterior region of lower jaw to be incisors, not canines, due to developmental status and position of teeth in alveolus. (B,C) Upper (B) and lower (C) dental arches of a 9.6-year-old Wolf-Hirschhorn girl (patient #5) who had a mixed dentition with permanent upper right central incisor and two lower incisors erupted. Dental enamel of the permanent teeth was hypoplastic (arrows). Two lower primary incisors were missing, and primary teeth were worn.

 

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