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PubMed1995

Amelogenesis imperfecta and nephrocalcinosis syndrome. Case studies of clinical features and ultrastructure of tooth enamel in two siblings.

This article describes the enamel ultrastructure and clinical features in two siblings with the little known syndrome of Amelogenesis imperfecta and nephrocalcinosis. Nephrocalcinosis was diagnosed by x-ray examination of the abdomen, intravenous pyelography, ultrasonography, and computed tomography scan. Amelogenesis imperfecta was diagnosed from clinical and histologic examinations. The affected enamel was hypoplastic (approximately 0.2 mm thick), positively birefringent, generally aprismatic, porous, and consisted of loosely packed, randomly orientated, thin (approximately 10 nm wide), ribbonlike crystals. The enamel surface was rough, extensively cracked, and covered with ovoid or globular protrusions. Observations showed that in this case hypoplasia, hypocalcification, or hypomaturation defects were present in the same tooth, indicating that both secretory and maturation phases may have been affected. The study suggested the possibility of an abnormality in interstitial matrix, which could lead to dystrophic calcification in the kidney and abnormal tooth enamel formation. It also suggested the possibility of involvement of two separate but closely linked genes.

Adolescent↗
PubMedDate not supplied

Abnormalities of the maxillary incisors in children with cleft lip and palate.

Dental anomalies of the maxillary anterior teeth were studied in seventy-seven children affected by unilateral and bilateral clefts of the lip and alveolar process, with or without involvement of the palate. As for the permanent lateral incisor in the cleft area, our results show that its congenital absence is the most frequent abnormality followed by anomalies in size and shape and supernumerary teeth. Enamel hypoplasia was found to affect the permanent central incisor on the cleft side more frequently. Early recognition of tooth abnormalities during the primary dentition phase for an interceptive treatment of potentially severe problems was emphasized.

Adolescent↗
PubMed2004

Role of the Notch signalling pathway in tooth morphogenesis.

Notch receptors are involved in cell fate decisions through the process of lateral inhibition or inductive signalling. Jagged2 belongs to the family of transmembrane proteins that serve as the ligands for Notch receptors. We have analysed the expression of the Jagged2 gene in developing mouse teeth. Jagged2 expression is restricted in inner enamel epithelial cells that give rise to the ameloblasts. We have also examined the role of Jagged2 in tooth development using mutant mice that lack the domain of the Jagged2 protein required for interaction with the Notch receptors (DSL domain). Homozygous mutant mice die after birth, exhibit abnormal tooth morphology and fusions between the palatal and mandibular shelves. These results demonstrate that Notch signalling plays an essential role in tooth development.

Ameloblasts↗
PubMed1996

Identification of the cell type origin of odontoma-like cell masses in microphthalmic (mi/mi) mice by in situ hybridization.

Tooth abnormalities occur in microphthalmic (mi/mi) mice. The elongated odontogenic epithelium is interrupted by unresorbed bone at the basal end of the mi/mi incisor, with the epithelium gathered into cell clusters. These clusters develop to odontoma-like masses. To identify the origin of the cell types of these odontoma-like masses, the localization of osteonectin (Osn), osteocalcin (Osc), osteopontin (Osp), matrix Gla protein (MGP) and amelogenin (Am) mRNA in the process of tooth development in mi/mi and +/+ mice was investigated by means of in situ hybridization. Decalcified mandibles of neonatal, 5-, 10-, 14-day-old mice were examined. Osn and Osc mRNA, which localized in osteoblasts and odontoblasts, were also detected in the cells of odontoma-like masses in mi/mi mice. The cells expressing these mRNA were short, columnar and odontoblast-like. Am mRNA was detected in ameloblasts. In mi/mi mice, Am mRNA was also detected in ameloblastic cell clusters, which were formed by the tall columnar cells in the odontoma-like masses. No apparent Osp mRNA expression was detected in the masses. These results indicated that even in odontogenic abnormal cells resulting from physical obstruction in mi/mi mice, the genes that are involved in normal tooth development were still expressed.

Animals↗
PubMed1999

Tooth position and speech--is there a relationship?

Although it is widely accepted that teeth play an important role in speech production, the relationship between tooth position and speech remains controversial. This review paper examines the relevant studies and discusses the difficulties of scientific investigation in this area. The ability of patients to adapt their speech to compensate for abnormal tooth position is recognized, but the mechanisms for this adaptation remain incompletely understood. The overall conclusion is that while certain dental irregularities show a relationship with speech disorders, this does not appear to correlate with the severity of the malocclusion. There is no definitive proof that alteration of tooth position can improve articulation disorders.

Adaptation, Physiological↗
PubMed2002

Factors affecting degeneration in human temporomandibular joints as assessed histologically.

The influence of sex, age, tooth loss, and articular disc position on temporomandibular joint (TMJ) degeneration was evaluated in specimens collected at autopsy from 15 women and 38 men ranging in age from 15 to 92 yr. The position of the articular discs was classified as normal or abnormal, tooth loss was both counted and categorized. Degenerative changes of the articular tissues were assessed histologically and quantified, taking into account both the severity of structural alterations and their extension along the articular surface. This was recorded separately in the condyle, disc, and temporal component, three latero-medial joint regions, and putative load-bearing and non-load-bearing surfaces. Analysis of covariance with repeated measures served for testing contributing factors. It showed that the effects of sex and the number of missing teeth were insignificant, whereas age up to about 55-60 yr and reduction of dental arch length proved to be the most important factors. Load-bearing seemed to play a significant role mainly at younger ages, and the effect of disc position was significant, when internal derangement was combined with reduction of dental arch length. Thus, rising severity of TMJ degenerative changes appears to be associated primarily with increasing age. In addition, it may also depend on mechanical factors, in particular loss of molar support and, to a minor degree, abnormal disc position.

Adolescent↗
PubMed1999

Embryonic and postnatal injections of bromodeoxyuridine produce age-dependent morphological and behavioral abnormalities.

The mitotic marker 5-bromodeoxyuridine (BrdU) was injected twice daily (60 mg/kg) into pregnant hooded rats on one of embryonic days (E) 11, 12, 13, 15, 17, or 21, or into rat pups on postnatal day (P) 10. The principal findings were the following: (1) BrdU exposure on E11 produces profound effects on body morphology, and animals must be fed a special diet because of chronic tooth abnormalities; (2) BrdU exposure at E17 or earlier produces a change in coat spotting pattern, the precise pattern varying with age; (3) BrdU exposure on E15 or earlier produces a reduction in both brain and body weight; (4) BrdU exposure on E17 or earlier reduces cortical thickness; (5) BrdU exposure on E11-E13 and at P10 reduces cerebellar size relative to cerebral size; (6) spatial learning is significantly affected after injections of BrdU at E11-E17, but the largest effect is on E17; (7) the deficit in spatial learning may be related in part to a reduction in visual acuity; and (8) skilled forelimb ability is most disrupted after BrdU exposure at E15 but is also impaired after injections on E13 or earlier. BrdU thus has teratological effects on body, brain, and behavior that vary with the developmental age of the fetus or infant.

Abnormalities, Drug-Induced↗
PubMed1995

Intravenous reproductive and developmental toxicity studies of cimadronate (YM175), a novel bisphosphonate, in rats and rabbits.

Cimadronate (YM175) is a novel bisphosphonate with potent inhibitory activity on bone resorption under development for the treatment of tumor-induced hypercalcemia, metastatic bone disease and osteoporosis. We conducted intravenous reproductive toxicity and teratology studies (Segment I, II and III) of this compound in rats and teratology study in rabbits. The test compound was dissolved in physiological saline, which was also given as the vehicle control. Rats were administered at a dosage of 0.06, 0.16 and 0.62 mg/kg/day in the male Segment I study. Dose levels in the other studies in rats including the female Segment I were 0.16, 0.31 and 0.62 mg/kg/day. In the Segment I study, no treatment-related abnormalities were observed in reproductive parameters or fetuses. In the Segment II study, slightly retarded fetal ossification was noted at 0.31 mg/kg/day or more, but the incidence of malformation did not increase. In the Segment III study, death of the dams and abnormal tooth growth of offspring were observed at 0.16 mg/kg/day or more. Further Segment III study showed that the no toxic effect level was 0.003 mg/kg/day. In the rabbit teratology study, dose levels were 0.01, 0.025 or 0.05 mg/kg/day. No toxic effects on pregnant females or their litters were observed at up to 0.05 mg/kg/day.

Abnormalities, Drug-Induced↗
PubMedDate not supplied

[Oral rehabilitation in dentinogenesis imperfecta. Report of a case].

We present a case of Amelogenesis imperfecta associated with Dentinogenesis imperfecta, affecting the primary dentition which is rehabilitated under general anesthesia. Dentinogenesis imperfecta is a tooth abnormality which presents clinical, radiological and histological characteristics, they should be recognized by the dentist who will determine the treatment depending on age and grade of affection. In the primary dentition we recommend the use of stainless steel crowns do to it's resistance and easy adaptation which will remain in the mouth until it's normal exfoliation.

Child, Preschool↗
PubMed2004

A systematic review of the consequences of premature birth on palatal morphology, dental occlusion, tooth-crown dimensions, and tooth maturity and eruption.

This systematic review addresses the question whether prematurity results in alteration of palatal morphology, dental occlusion, tooth-crown dimensions, and tooth maturation. A literature survey from the PubMed database covering the period from January 1966 to November 2002 used the Medical Subject Headings terms "infant, premature," and "infant, low birth weight" in combination with "jaws," "dental physiology," "dentition," and "tooth abnormalities." Controlled studies written in English and with definitions of premature birth according to the World Health Organization were selected. Two reviewers selected and extracted the data independently and also assessed the quality of the studies. The search strategy resulted in 113 articles, of which 13 met the inclusion criteria. Scientific evidence was found for altered palatal morphology in the short term among the premature children, and oral intubation was a contributing factor to the alterations. If corrected age was considered for the premature children, no delay in dental development and eruption was found compared with normally born children. Thus, the early birth of premature children must be taken in account when planning for orthodontic treatment. Because of the contradictory results and lack of longitudinal studies, the scientific evidence was too weak to answer the questions whether premature birth causes permanent alteration of palatal morphology, alteration of dental occlusion, and altered tooth-crown dimensions. To answer these questions and obtain reliable scientific evidence whether premature children are at risk for malocclusions from possible alterations of palatal morphology such as asymmetry and high arched palates, further well-designed controlled studies as well as longitudinal studies are needed.

Dental Research↗
PubMed2002

Lunatic fringe, FGF, and BMP regulate the Notch pathway during epithelial morphogenesis of teeth.

Teeth develop as epithelial appendages, and their morphogenesis is regulated by epithelial-mesenchymal interactions and conserved signaling pathways common to many developmental processes. A key event during tooth morphogenesis is the transition from bud to cap stage when the epithelial bud is divided into specific compartments distinguished by morphology as well as gene expression patterns. The enamel knot, a signaling center, forms and regulates the shape and size of the tooth. Mesenchymal signals are necessary for epithelial patterning and for the formation and maintenance of the epithelial compartments. We studied the expression of Notch pathway molecules during the bud-to-cap stage transition of the developing mouse tooth. Lunatic fringe expression was restricted to the epithelium, where it formed a boundary flanking the enamel knot. The Lunatic fringe expression domains overlapped only partly with the expression of Notch1 and Notch2, which were coexpressed with Hes1. We examined the regulation of Lunatic fringe and Hes1 in cultured explants of dental epithelium. The expression of Lunatic fringe and Hes1 depended on mesenchymal signals and both were positively regulated by FGF-10. BMP-4 antagonized the stimulatory effect of FGF-10 on Lunatic fringe expression but had a synergistic effect with FGF-10 on Hes1 expression. Recombinant Lunatic fringe protein induced Hes1 expression in the dental epithelium, suggesting that Lunatic fringe can act also extracellularly. Lunatic fringe mutant mice did not reveal tooth abnormalities, and no changes were observed in the expression patterns of other Fringe genes. We conclude that Lunatic fringe may play a role in boundary formation of the enamel knot and that Notch-signaling in the dental epithelium is regulated by mesenchymal FGFs and BMP.

Animals↗
PubMed1998

Oral manifestations of hereditary sensory and autonomic neuropathy type IV. Congenital insensitivity to pain with anhidrosis.

OBJECTIVE: Hereditary sensory and autonomic neuropathy type IV (congenital insensitivity to pain with anhidrosis) is a rare disorder. In this study, we investigated the oral and dental manifestations associated with hereditary sensory and autonomic neuropathy type IV. STUDY DESIGN: Eighteen patients with hereditary sensory and autonomic neuropathy type IV whose ages ranged from 1 year 0 months to 22 years 3 months were examined for oral signs and symptoms of tooth abnormalities, malocclusions, soft tissue disorders, tongue papilla atrophy, and morphologic abnormalities of hands and fingers. RESULTS: All 18 patients showed congenital insensitivity to pain and anhidrosis. Oral self-mutilations, such as autoextraction of teeth and severe biting injuries (with resultant scarring) of the finger tips and oral soft tissues (tongue, lip, and buccal mucosa), were found in most patients. In infant patients the condition was typically characterized by decubital ulcers on the ventral surface of the tongue, resulting from trauma of the incisal edge of erupting mandibular primary incisors during sucking or nursing. These ulcers led to several local and systemic problems, such as tongue bleeding, infection, malnutrition, and halitosis. A large number of missing teeth and a high incidence of dental caries were additional characteristic findings. Such oral self-mutilations were found to decrease with age and with the intellectual, social, and/or emotional development of the patients. However, not all of the mutilations were completely eliminated. Two patients had partial dentures to replace missing teeth. CONCLUSIONS: Our study suggests that early diagnosis and specific dental management for patients with hereditary sensory and autonomic neuropathy type IV are important for prevention of the characteristic oral and dental problems accompanying this disorder.

Adolescent↗
PubMed1990

New cases of dermoodontodysplasia?

We report on 2 sisters and one brother with severe dental anomalies, trichodysplasia, onychodysplasia, and slight skin alterations. Four other relatives have only mild dental anomalies. Differential diagnosis includes 3 other ectodermal dysplasias: hypodontia and nail dysgenesis, dermoodontodysplasia, and trichodermodysplasia with dental alterations. Cause is unknown.

Abnormalities, Multiple↗
PubMed2003

Multiple macrodontic multituberculism.

Ekman-Westborg and Julin [1974: Oral Surg 38:217-222], described multiple macrodontia and multituberculism affecting the teeth without other anomalies (E-WJ). We describe a Chilean case in a 12-year-old with the typical dental alterations and with histopathologic findings that include absence of predentin layer and prominent reduced enamel epithelium. E-WJ is not a syndrome and we propose "multiple macrodontic multituberculism" as a better name for this anomaly of uncertain etiology affecting only the crowns of the teeth.

Bicuspid↗
PubMed2003

Analysis of colored teeth from Precolumbian Tlatelolco: postmortem transformation or intravitam processes?

The etiological basis of the abnormal coloration of archaeological teeth has been an unsolved question for a long time. Differences in the appearance of some archaeological teeth from Precolumbian adult and infant skeletons, detected by external optical inspection, led us to study this problem. A blue stain is visible in a few of the deciduous erupted teeth, and a brown color in various unerupted teeth in the collection, while brown spots appear on some permanent teeth. Several processes or factors that may occur during one's life, others around the time of death, and still others resulting from postmortem alterations have been reported as potential causes of abnormal tooth coloration.A sample of 35 colored teeth and two soil layers taken from Tlatelolco were analyzed by particle-induced X-ray emission (PIXE) as well as selective dissolution techniques. Concentrations of total and extractable elements in enamel and soil layers (Cg1-Cg2) were obtained. This paper describes the occurrence and implications of a substantial secondary concentration of Zinc (Zn), manganese (Mn), strontium (Sr), and iron (Fe) in the deciduous erupted and nonerupted teeth as compared to that in the adult teeth. Our interpretation is that, in this archaeological context, the brown spots and blue stains on the teeth are due to differences in tooth enamel porosity and to a postmortem biogeochemical process. The alterations involve cumulization and diagenesis of iron, manganese, and organic matter solutions that were eluviated from the soil and are not the result of antemortem or perimortem conditions such as trauma or disease.

Color↗