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Hypocalcified type of amelogenesis imperfecta in a large family: clinical, radiographic, and histological findings, associated dento-facial anomalies, and resulting treatment load.
Acta Odontol Scand. 2009; 67(4):240-7.AO

Abstract

OBJECTIVE

The purpose of this study was to report on the clinical, radiographic, and histological dental findings and the resulting treatment load in a five-generation family with amelogenesis imperfecta (AI).

MATERIAL AND METHODS

Thirteen affected and 15 unaffected individuals were examined clinically and radiographically. In addition, four exfoliated deciduous teeth were examined by scanning electron microscopy and microradiography.

RESULTS

The mode of inheritance of AI was autosomal-dominant. At eruption, most of the tooth enamel was yellow, lacking translucency, and prone to gradual loss in subjects with AI. Post-eruptive breakdown of enamel was extensive in accordance with the histological observations of hypomineralized and porous enamel. Extensive enamel loss and discoloration were observed in older affected individuals. The treatment need had been extensive: 76.2% of the total number of teeth present in affected individuals had been treated with partial or full coverage compared to 1.7% of the teeth in unaffected relatives. Unaffected individuals had more endodontically treated teeth than AI-affected relatives. Adjunctive findings, e.g. tooth agenesis, tooth impaction, pulp stones, enlarged follicular space, and taurodontism, were rare in both groups.

CONCLUSIONS

Affected family members had the hypocalcified type of AI, which is characterized by severe hypomineralization, extensive post-eruptive loss, and discoloration of the enamel. Adjunctive findings were rare. Individuals with the hypocalcified type of AI have an extensive restorative treatment load compared to unaffected relatives.

Authors+Show Affiliations

The Center for Oral Health in Rare Medical Conditions, Denmark.No affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Comparative Study
Journal Article
Research Support, Non-U.S. Gov't

Language

eng

PubMed ID

19452331

Citation

Gjørup, Hans, et al. "Hypocalcified Type of Amelogenesis Imperfecta in a Large Family: Clinical, Radiographic, and Histological Findings, Associated Dento-facial Anomalies, and Resulting Treatment Load." Acta Odontologica Scandinavica, vol. 67, no. 4, 2009, pp. 240-7.
Gjørup H, Haubek D, Hintze H, et al. Hypocalcified type of amelogenesis imperfecta in a large family: clinical, radiographic, and histological findings, associated dento-facial anomalies, and resulting treatment load. Acta Odontol Scand. 2009;67(4):240-7.
Gjørup, H., Haubek, D., Hintze, H., Haukali, G., Løvschall, H., Hertz, J. M., & Poulsen, S. (2009). Hypocalcified type of amelogenesis imperfecta in a large family: clinical, radiographic, and histological findings, associated dento-facial anomalies, and resulting treatment load. Acta Odontologica Scandinavica, 67(4), 240-7. https://doi.org/10.1080/00016350902973685
Gjørup H, et al. Hypocalcified Type of Amelogenesis Imperfecta in a Large Family: Clinical, Radiographic, and Histological Findings, Associated Dento-facial Anomalies, and Resulting Treatment Load. Acta Odontol Scand. 2009;67(4):240-7. PubMed PMID: 19452331.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Hypocalcified type of amelogenesis imperfecta in a large family: clinical, radiographic, and histological findings, associated dento-facial anomalies, and resulting treatment load. AU - Gjørup,Hans, AU - Haubek,Dorte, AU - Hintze,Hanne, AU - Haukali,Gro, AU - Løvschall,Henrik, AU - Hertz,Jens Michael, AU - Poulsen,Sven, PY - 2009/5/20/entrez PY - 2009/5/20/pubmed PY - 2012/9/26/medline SP - 240 EP - 7 JF - Acta odontologica Scandinavica JO - Acta Odontol Scand VL - 67 IS - 4 N2 - OBJECTIVE: The purpose of this study was to report on the clinical, radiographic, and histological dental findings and the resulting treatment load in a five-generation family with amelogenesis imperfecta (AI). MATERIAL AND METHODS: Thirteen affected and 15 unaffected individuals were examined clinically and radiographically. In addition, four exfoliated deciduous teeth were examined by scanning electron microscopy and microradiography. RESULTS: The mode of inheritance of AI was autosomal-dominant. At eruption, most of the tooth enamel was yellow, lacking translucency, and prone to gradual loss in subjects with AI. Post-eruptive breakdown of enamel was extensive in accordance with the histological observations of hypomineralized and porous enamel. Extensive enamel loss and discoloration were observed in older affected individuals. The treatment need had been extensive: 76.2% of the total number of teeth present in affected individuals had been treated with partial or full coverage compared to 1.7% of the teeth in unaffected relatives. Unaffected individuals had more endodontically treated teeth than AI-affected relatives. Adjunctive findings, e.g. tooth agenesis, tooth impaction, pulp stones, enlarged follicular space, and taurodontism, were rare in both groups. CONCLUSIONS: Affected family members had the hypocalcified type of AI, which is characterized by severe hypomineralization, extensive post-eruptive loss, and discoloration of the enamel. Adjunctive findings were rare. Individuals with the hypocalcified type of AI have an extensive restorative treatment load compared to unaffected relatives. SN - 1502-3850 UR - https://www.unboundmedicine.com/medline/citation/19452331/Hypocalcified_type_of_amelogenesis_imperfecta_in_a_large_family:_clinical_radiographic_and_histological_findings_associated_dento_facial_anomalies_and_resulting_treatment_load_ L2 - https://www.tandfonline.com/doi/full/10.1080/00016350902973685 DB - PRIME DP - Unbound Medicine ER -