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PubMed2000

Amelogenesis imperfecta: functional and esthetic restoration of a severely compromised dentition.

The treatment of patients with severe forms of amelogenesis imperfecta presents an interesting challenge to the dental team. A 16-year-old girl presented with a severely compromised occlusion and poor esthetics that had lowered her self-esteem. Preoperative investigations included the determination of completion of growth, an accurate diagnostic waxup, and an evaluation of clinical crown lengths. Periodontal full-flap surgery was planned to lengthen clinical crowns and create gingival harmony. This was to be followed by placement of 28 complete crowns to restore occlusal function and gain a pleasing esthetic result. All-porcelain crowns were placed from premolar to premolar in each arch (20 crowns), and porcelain-fused-to-metal crowns were placed on each molar tooth in each arch (8 crowns). The final treatment result provided this patient with a mutually protective occlusion and with esthetics that greatly enhanced her self-image.

Adolescent↗
PubMed1993

Taurodontism of the mandibular first permanent molar distinguishes between the tricho-dento-osseous (TDO) syndrome and amelogenesis imperfecta.

The diagnosis of tricho-dento-osseous (TDO) syndrome is often confused with that of a variant of amelogenesis imperfecta (AI) which shows similar dental features of hypomaturation-hypoplastic enamel defects and putative taurodontism. In this controlled study, an objective, biometric measurement technique was used to determine the prevalence and severity of taurodontism of the mandibular first permanent molar in 23 patients with AI and one patient with TDO syndrome compared with age- and sex-matched controls. The published radiographs of previous cases of TDO and hypomaturation-hypoplastic AI were also reviewed with regard to the presence and severity of taurodontism. The results indicate that in all cases of TDO, taurodontism of the molars including mandibular first permanent molars was consistently present and in a severe form. By contrast, the taurodontic defects present in all cases of AI, including the hypomaturation-hypoplastic variant were not significantly different from matched, healthy controls. Of significance is the fact that in all the AI cases, none of the taurodontic defects were present on the mandibular first permanent molars. The results indicate that true taurodontism as indicated by a change in the mandibular first permanent molars occurs only in the TDO syndrome. This feature may be used to differentiate clearly between TDO and AI.

Abnormalities, Multiple↗
PubMed2003

Evaluation of oral and systemic manifestations in an amelogenesis imperfecta population.

OBJECTIVES: The aim of this investigation was to describe the dental and craniofacial characteristics of patients with amelogenesis imperfecta (AI). METHODS: The study group included 43 patients(33 female and 10 male) with a mean age of 11.4+/-2.6 years. A panoramic and a cephalometric radiograph were obtained from each of these patients. Clinically AI cases were divided into four main groups according to Witkop. All patients were evaluated for chronological, bone and dental age. The patients who had severe retarded bone age were evaluated for plasma growth hormone(GH) concentrations. RESULTS: Dental and bone ages were retarded with respect to chronological age in five patients. Dental maturity and tooth eruption were not age- appropriate in some of our patients. In type III AI patients a delay in skeletal age was observed. Severe late eruption was seen in 3 patients, severe delay in dental maturity was noted in patients with type IV AI. Dental age was clinically lower in GH-deficient subjects, and skeletal age was consistently more retarded than dental age when compared to chronological age. Anterior open bite was present in both primary and permanent dentitions of 50% of the patients with type I AI, 30.8% of the patients with type II AI, and 60% of type III AI. CONCLUSION: It is concluded that the primary structure for the classification of AI be based on the mode of inheritance, with the clinical and radiographic appearances (and any other features such as systemic findings) being the secondary discriminators.

Age Determination by Skeleton↗
PubMed2003

Restoring function and esthetics in a patient with amelogenesis imperfecta: a clinical report.

This clinical report describes the prosthodontic treatment for an 18-year-old man diagnosed with amelogenesis imperfecta. The aim of treatment was to reduce dental sensitivity and to restore esthetics and masticatory function. Metal-ceramic fixed partial dentures were placed on posterior teeth to modify the occlusion, and porcelain laminate veneers were placed to improve the esthetics of the maxillary anterior teeth. Clinical examination 12 months after treatment revealed no evidence of disorders associated with the restored teeth or their supporting structures.

Adolescent↗
PubMed1995

Amelogenesis imperfecta in triplets: a unique family record.

A unique family is recorded in which triplets were observed with the hypomaturation type of amelogenesis imperfecta. A simple technique of DNA analysis showed that two of the more severely affected triplets were monozygotic and the third less severely affected triplet dizygotic.

Alleles↗
PubMed2004

An interdisciplinary approach for restoring function and esthetics in a patient with amelogenesis imperfecta and malocclusion: a clinical report.

This clinical report describes an interdisciplinary approach for the coordinated treatment of a patient diagnosed with amelogenesis imperfecta and malocclusion. The patient's functional and esthetic expectations were successfully met with interdisciplinary treatments, including orthodontics, porcelain laminate veneers, metal-ceramic fixed partial dentures, and direct composite restorations.

Adult↗
PubMed1996

A new form of skeletal dysplasia with amelogenesis imperfecta and platyspondyly.

We report two patients, born of consanguineous parents, affected by a disorder resulting in mild growth retardation. Hallmarks are amelogenesis imperfecta (absence of the enamel cap) associated with brachyolmia-like anomalies: platyspondyly with short pedicles, narrow intervertebral and interpedicular distances, rectangular-shaped vertebrae with posterior scalloping and herniation of the nuclei, and broad femoral necks. Inheritance appears to be autosomal recessive.

Abnormalities, Multiple↗
PubMed1986

Amelogenesis imperfecta: prevalence and incidence in a northern Swedish county.

The aim of the study was to establish the prevalence and incidence of amelogenesis imperfecta (AI) in the county of Västerbotten, northern Sweden using the criteria of Witkop & Sauk (1976). The diagnoses were based upon clinical and radiographic evidence. The study-population consisted of all 3-19-yr-olds born in the county from 1963 to 1979. AI was diagnosed in 79 children, 41 girls and 38 boys, giving a prevalence of 1.4:1000. The mean incidence 1963-79 was 1.3:1000. Half of the children with AI had brothers or sisters in the group. The hypoplastic form was the most common (58 children) followed by the hypomaturation form (16 children). Only five children had the hypomineralization form.

Adolescent↗
PubMed2001

Amelogenesis imperfecta: diagnosis and resolution of a case with hypoplasia and hypocalcification of enamel, dental agenesis, and skeletal open bite.

A case of amelogenesis imperfecta with hypoplasia, hypocalcification of the enamel, congenital absence of teeth 12 and 22, delayed eruption of tooth 23, edge-to-edge incisal relationship, open bite, and bilateral posterior cross bite at the level of the first and second premolars is presented. Lateral skull teleradiography indicated a Class III skeletal pattern of maxillary origin associated with a dolichofacial pattern with multiple indicators of facial hyperdivergence. The patient presented a major esthetic abnormality of the face and required orthodontic treatment prior to a prosthetic solution with full-coverage metal-ceramic crowns in both the maxilla and the mandible. The diagnosis of cases such as this one and the therapeutic implications from an orthodontic and prosthetic standpoint are reported.

Adolescent↗
PubMed1998

Open bite deformity in amelogenesis imperfecta. Part 1: An analysis of contributory factors and implications for treatment.

Anterior open bite (AOB) is often seen in patients with amelogenesis imperfecta (AI). The skeletal and dental components were analysed in 15 patients with AI and AOB. Measurements on cephalometric radiographs and dental models were compared with those of 130 patients with AOB but without enamel anomalies. Skeletal components in the AI and non-AI group were comparable. The AI group showed omega-shaped dental arches and a reversed mandibular curve of Spee. Orthodontic treatment options are limited because of the conical form of the teeth, tight contacts in the posterior regions and the condition of enamel resulting in difficulties in bonding brackets. Fixation problems encountered during surgery are discussed. A multidisciplinary treatment is outlined consisting of a multi-segment Le Fort I osteotomy followed by prosthetic rehabilitation to stabilize occlusion for at least one year postoperatively.

Adolescent↗
PubMed1995

The mineral and protein content of enamel in amelogenesis imperfecta.

The purpose of this investigation was to characterize the enamel composition of teeth affected with the hereditary enamel disorders known as amelogenesis imperfecta. Teeth from 10 individuals representing all 3 major AI types (hypocalcified, n = 3; hypomaturation, n = 3; hypoplastic, n = 4) and 10 normal teeth were studied. Half of each tooth was used for histological and biochemical studies. The enamel protein content was estimated by amino acid analysis. The enamel mineral content (volume %) was determined from the calcium and/or phosphorus content. Calcium was measured using atomic absorption and phosphorus was determined colorimetrically. The mean enamel mineral content was reduced for all hypomaturation and hypocalcified AI teeth while hypoplastic AI enamel varied from normal to reduced compared with normal enamel. The enamel protein content was increased in all but one AI case (7 cases were examined for protein) compared with the normal enamel. The mineral and protein content in AI enamel showed a significant inverse relationship (R = -0.939, P = 0.001). This study shows that all three of the major AI groups can have subtypes associated with substantial decreases in the enamel mineral content, although hypomineralization appears most severe in the hypomaturation and hypocalcified AI types. The decreased mineral content was associated with an increased protein content in AI enamel. These findings provide further evidence that altered enamel mineralization in AI teeth likely involves abnormal post-secretory processing of the enamel proteins.

Amelogenesis Imperfecta↗
PubMed1985

Interradicular dentin dysplasia associated with amelogenesis imperfecta.

Very few reports are available on the occurrence of dysplasia of both enamel and dentin. This report concerns a rare association of amelogenesis imperfecta with a dysplasia of dentin in the interradicular area in sisters of Japanese descent who have no other morphologic anomalies. Retarded tooth eruption was also a clinical feature in both sisters. Histologic examination of several teeth revealed that the anomalous interradicular dentin consisted of a mass of small, onion-like calcified bodies. The absence of any dental abnormalities in both parents, who are related as first cousins, supports the concept of autosomal recessive inheritance for this trait.

Adolescent↗
PubMed1996

Local, hypoplastic type of amelogenesis imperfecta: a clinical, genetic, radiological and dermatoglyphic study.

A patient and her family members showing X-linked dominant form local, hypoplastic type of amelogenesis imperfecta (AI) were investigated from view-point of their teeth, clinical, genetic, radiological and particularly dermatoglyphic findings. It was suggested that it might be a close relationship between the intra-uterine development of both AI and unusual dermatoglyphs originated from the same layer, ectoderm and an X-linked dominant gene could determine both abnormal teeth and unusual dermatoglyphic characteristics in questions.

Adolescent↗
PubMed1994

Mineral distribution in the enamel of teeth with amelogenesis imperfecta as determined by quantitative microradiography.

The aim of the present investigation was to determine the mineral distribution in the enamel of teeth with amelogenesis imperfecta (AI) by quantitative microradiography. A further aim was to correlate the findings to clinical manifestations and inheritance patterns for AI. Included in the study were a total of 29 teeth with AI, 28 primary and one permanent, and seven unaffected teeth, five primary and two permanent. Quantitative microradiography was applied to sagittally ground sections, 70-90 microns, of the teeth. The mineral content of the enamel, expressed as percentage by volume, was lower in most of the teeth with AI than in the unaffected teeth. The largest range for the mineral distribution was found in the enamel of the variants clinically characterized by hypomineralization. These teeth showed a mineral distribution pattern that reflected an extremely low mineral content in the bulk of the enamel. In the AI teeth clinically characterized by hypoplasia, the mineral distribution pattern was similar to that of the unaffected teeth, although with larger local variations in mineral content. Apart from the teeth connected with X-linked inheritance, no differences were found among teeth with similar clinical variants connected with different inheritance patterns.

Amelogenesis Imperfecta↗
PubMed1986

Hereditary aspects and classification of hereditary amelogenesis imperfecta.

In an epidemiologic study in the middle of Sweden comprising 425 000 children age 3-19 yr, 105 children were diagnosed as having hereditary amelogenesis imperfecta (HAI). The material then was primarily classified upon clinical criteria into different subgroups either associated to a hypoplastic or to a hypomineralized type of HAI. Analyzing available genetic data obtained from 95 children in 76 families and further 11 adults in 10 families, eight different entities of HAI could be identified in a classification of HAI. In both of the basic types of HAI, the hypoplastic and the hypomineralized, autosomal dominance was the most common mode of inheritance, even if an autosomal recessive inheritance could be identified in some of the subgroups of HAI. The hypoplastic type (rough-pitted), autosomal dominance with incomplete penetrance was dominating in the material, 47 out of 105 cases. The clinical classification seemed to be relevant when testing the material on a genetic basis showing that the clinical criteria provide a strong support for a classification of HAI.

Adolescent↗