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Two male siblings are described with a clinical and molecular diagnosis of X-linked Opitz (G/BBB) syndrome and the previously unreported feature of neonatal mandibular incisors.
A 2-year-and-6-month-old Japanese girl with delayed eruption of a maxillary right primary canine and disturbance in the development of maxillary right primary molars was examined. The crown of the maxillary right first primary molar was severely decayed and hypoplastic. The periodontal tissues on the maxillary right posterior region were swollen and slight erythrogenic change was observed. Radiographic examination revealed a disturbance in the development of the permanent successor and the primary teeth in the maxillary right quadrant. According to the history, the maxillary right first primary molar had erupted on day seven after birth. It is suggested that the disturbance in development of the permanent successor and the primary teeth was secondary to osteitis caused by infection of the neonatal tooth. Appropriate dental treatment had not been administered for more than two years because the neonatal molar had not been diagnosed.
OBJECTIVE: The aim of the present study was to estimate the prevalence of dental fluorosis in primary dentition of a San Luis Potosi children population, and its association to fluoride concentration in drinking water and urine. An additional objective was, to develop, validate, and test a specific index for dental fluorosis in primary dentition. MATERIAL AND METHODS: From May 1997, to January 1999, we conducted a cross-sectional study to assess the prevalence of dental fluorosis in primary dentition. Study subjects were 100 children aged 3-6 years, selected at random from three kindergartens in three risk areas of San Luis Potosi. The specific index of dental fluorosis for primary dentition (Dental Fluorosis for Primary Dentition Index--DFPDI) was validated by estimating fluoride concentrations in enamel of teeth with and without dental fluorosis. The Kruskal-Wallis test was used to assess the association between fluoride concentrations in drinking water and urine, with dental fluorosis; the association between risk area and dental fluorosis was assessed with the Mantel-Haenszel chi 2 test. RESULTS: The prevalence of dental fluorosis in primary dentition was 78%; primary molars were most affected in both maxillae and the predominant color was a non-glossy white appearance. We found a strong direct correlation (r = 0.93) between fluoride concentrations in primary teeth and the DFPDI. Associations were found between fluoride concentrations of drinking water and urine, with dental fluorosis (Kruskal-Wallis p = 0.00001), and between risk area and dental fluorosis (Mantel-Haenszel chi 2 p = 0.00001). CONCLUSIONS: DFPDI allowed adequate identification and grading of dental fluorosis in primary dentition. It is important to detect the initial toxic effects of fluoride exposure to predict dental fluorosis in permanent dentition and skeletal fluorosis.
The term Riga-Fede disease has been used historically to describe traumatic ulceration that occurs on the ventral surface of tongue in neonates and infants. It is most often associated with natal and neonatal teeth in newborns. A case of Riga-Fede disease is presented.
Teeth have been missing from birds (Aves) for at least 60 million years. However, in the chick oral cavity a rudiment forms that resembles the lamina stage of the mammalian molar tooth germ. We have addressed the molecular basis for this secondary loss of tooth formation in Aves by analyzing in chick embryos the status of molecular pathways known to regulate mouse tooth development. Similar to the mouse dental lamina, expression of Fgf8, Pitx2, Barx1, and Pax9 defines a potential chick odontogenic region. However, the expression of three molecules involved in tooth initiation, Bmp4, Msx1, and Msx2, are absent from the presumptive chick dental lamina. In chick mandibles, exogenous bone morphogenetic protein (BMP) induces Msx expression and together with fibroblast growth factor promotes the development of Sonic hedgehog expressing epithelial structures. Distinct epithelial appendages also were induced when chick mandibular epithelium was recombined with a tissue source of BMPs and fibroblast growth factors, chick skin mesenchyme. These results show that, although latent, the early signaling pathways involved in odontogenesis remain inducible in Aves and suggest that loss of odontogenic Bmp4 expression may be responsible for the early arrest of tooth development in living birds.
Restrictive dermopathy (RD) is a lethal autosomal recessive genodermatosis (MIM No. 275210) in which tautness of the skin causes fetal akinesia or hypokinesia deformation sequence (FADS). Polyhydramnios with reduced fetal movements is followed by premature delivery at around 31 weeks gestation. Manifestations include a tightly adherent, thin, translucent skin with prominent vessels, typical facial changes, generalized joint contractures, enlarged fontanelles, dysplasia of clavicles, respiratory insufficiency, and an enlarged placenta with short umbilical cord. Histologic abnormalities of the skin include thin dermis with paucity and hypoplasia of the appendages and abnormally arranged collagen bundles. Elastic fibers are nearly missing. The subcutaneous fat is slightly increased. These skin findings usually appear after 22 or 24 weeks of gestation, which is why prenatal diagnosis with skin biopsy may fail. This disease is easily differentiated from other congenital FADS, such as Pena-Shokeir syndrome, COFS syndrome, Parana hard-skin syndrome, etc. We report on an affected boy of consanguineous parents and 30 previous cases are reviewed.
The pediatric nurse practitioner advises families in proper oral hygiene, fluoride supplementation, prevention of damaging oral habits and tooth decay, and the need for children to see a pediatric dentist. This article presents the issues of oral health for children from infancy through adolescence. Common pathologic conditions, oral trauma, and the use of sealants in children are also described.
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A Japanese girl was referred to Osaka University Dental Hospital for examination of a tooth-like structure that had erupted following spontaneous exfoliation of a natal tooth in the lower left primary central incisor region. The structure had erupted at 6 months of age, and radiographic and clinical examination showed composition of pulp and dentin, but no enamel. On histological examination, the majority of the dentin area had a tubular dentin-like appearance, while the outer area of the root appeared to be composed of an osteodentin-like substance. Most of the dentin was covered by cementum. These findings suggest that the structure had originated from a developing remnant of the extracted natal tooth, which must have remained in the gingival tissues. We termed this calcified structure a residual natal tooth.
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In this report we present the clinical history and findings in a female newborn with 4q deficiency/14q duplication, the unbalanced product of a paternal t(4;14)(q33;q32). The clinical symptoms and signs observed in this child up to the age of 14 months were most compatible with the diagnosis of Hallermann-Streiff syndrome.
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Self-mutilation of tongue is a type of self-injurious behavior. Ulcers of the lingual frenum in neonates with natal lower incisors are referred to as Riga-Fede disease. In this paper a case of Riga-Fede disease in a ten-month infant male with lower central incisors is reported. The ulcer resolved after the sharp incisal edges were smoothened and topical triameinolone was applied. As this lesion may be confused or associated with other serious disorders, a review of medical and dental literature was included.
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The 'problem' of the (neo)natal tooth is discussed in conjunction with a report of two cases and a review of the literature. The estimated incidence is between 1:700 and 1:3500 births. Girls are more often affected than boys. It usually concerns the two lower central deciduous incisors. The cause is most likely to be a superficial position of the tooth-germ, in which a hereditary component is a positive factor. The tooth is often deficient and very mobile, which may require treatment. In these cases extraction is the best therapy, if necessary after administration of vitamin K.
As an introduction, this article describes the circumstances under which dental anatomy was taught to first year students at Utrecht University on November 26, 1945, as this led to the observation of a dens natalis. Relevant literature is discussed concerning the significance of the dens natalis for the child's future and for the family. Investigations into the frequency of the phenomenon are mentioned. Variations of the phenomenon are discussed in two cases. An overview of feeding and eating difficulties and measures to be taken concludes the article.
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