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Int J Paediatr Dent. 2008 Jul;18(4):306-11. doi: 10.1111/j.1365-263X.2007.00907.x. Epub 2008 Mar 6.

Solitary median maxillary central incisor syndrome occurring together with oromandibular-limb hypogenesis syndrome type 1: a case report of this previously unreported combination of syndromes.

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  • 1Faculty of Dentistry, Khon Kaen University, Khon Kaen, Thailand.



Solitary median maxillary central incisor syndrome is a unique developmental abnormality, involving the central incisor tooth germs, occurring with and without systemic involvement. This syndrome has been recorded in association with many other midline developmental anomalies and several known syndromes. Its presence, together with oromandibular-limb hypogenesis syndrome type 1, has not previously been reported.


A 3-year-old girl was presented with early childhood caries and a solitary median maxillary central incisor. She had a median submucosal cleft palate and severe micrognathia with hypoglossia. In addition, she had short stature, eating difficulty, and hearing and speech problems. Comprehensive dental treatment for severe early childhood caries was accomplished with a 3-month follow-up appointment to monitor the oral health. The multidisciplinary consultation important for further management has been established.


The results suggested that when a solitary median maxillary incisor tooth presents, a paediatrician and a geneticist should be asked to carefully examine the patient for other craniofacial malformations and especially midline systemic problems.

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