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BMC Oral Health
Published in: BMC Oral Health 1/2016

Open Access 01-12-2016 | Case report

Current concepts in odontohypophosphatasia form of hypophosphatasia and report of two cases

Authors: Zhu-yu Wang, Kai Zhang, Guang-sen Zheng, Wei Qiao, Yu-xiong Su

Published in: BMC Oral Health | Issue 1/2016

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Abstract

Background

Hypophosphatasia is a rare inherited disease derived from mutations in tissue non-specific alkaline phosphatase genes, with typical oral symptoms including short root anomaly and dysplasia of dentin or cementum.

Case presentation

Two young female patients presented with short root anomaly with a history of premature loss of deciduous and/or permanent teeth. The laboratory and imaging investigations were performed. One case was diagnosed as odontohypophosphatasia concurrent with hyperthyroidism, the other was odontohypophosphatasia concurrent with multiple radicular cysts.

Conclusion

This report presents two cases of odontohypophosphatasia, a rare disease which is difficult to be diagnosed, and highlights that the history of premature loss of deciduous and/or permanent teeth, oral manifestation and laboratory tests are crucial for clinical diagnosis.
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Metadata
Title
Current concepts in odontohypophosphatasia form of hypophosphatasia and report of two cases
Authors
Zhu-yu Wang
Kai Zhang
Guang-sen Zheng
Wei Qiao
Yu-xiong Su
Publication date
01-12-2016
Publisher
BioMed Central
Published in
BMC Oral Health / Issue 1/2016
Electronic ISSN: 1472-6831
DOI
https://doi.org/10.1186/s12903-016-0266-0

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