Dentinogenesis imperfecta in a racial isolate with multiple hereditary defects

Hursey, Rudolph J.; Witkop, Carl J.; Miklashek, Doris; Sackett, Lee M.

doi:10.1016/0030-4220(56)90325-5

Cited by 37 publications

(10 citation statements)

References 12 publications

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“…Enamel hypoplasias have been described in connection with DI [29]. The occurrence of these aberrations in enamel in OI has been regarded as a secondary effect of the defective dentine.…”

Section: Discussionmentioning

confidence: 99%

Morphology of dental enamel and dentine–enamel junction in osteogenesis imperfecta

Lindau

Dietz

et al. 1999

Int J Paed Dentistry

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“…Enamel hypoplasias have been described in connection with DI [29]. The occurrence of these aberrations in enamel in OI has been regarded as a secondary effect of the defective dentine.…”

Section: Discussionmentioning

confidence: 99%

Morphology of dental enamel and dentine–enamel junction in osteogenesis imperfecta

Lindau

Dietz

et al. 1999

Int J Paed Dentistry

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“…Thus, types I and I1 DI have been suggested to share a common disturbance in the differentiation of odontoblasts [Takagi and Sasaki, 1988;Sauk, 19901. Qpe I11 Separate type I11 DI was first described in a triracial isolate in Brandywine, Maryland [Hursey et al, 1956;Witkop et al, 19661. The clinical features closely resemble those of type I1 DI.…”

Section: Dentinogenesis Imperfecta Vpes I and I1mentioning

confidence: 99%

Heritable dentin defects: Nosology, pathology, and treatment

1993

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Heritable dentin defects have been divided into 2 main categories: dentinogenesis imperfecta (DI) and dentin dysplasia (DD). Recent studies have shown that they share many features in common. Of the connective tissue diseases, only osteogenesis imperfecta (OI) has been linked to these disorders. So far, no definitive relation between the type of OI and the dental involvement can be established. Familial occurrence of DI with OI cannot be comprehensively explained by mutations in type I collagen genes. No information about the gene defects in DD is available. At the ultrastructural level, the organization of the normally cross-striated collagen fibers in the dentin matrix varies markedly in patients affected by DI.

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“…Pulp chambers of permanent teeth are obliterated and areas of rarefaction, which may not be attributed to caries or attrition, may occur at their apices. Anterior open bites were found in affected individuals .…”

Section: Classification Of Hereditary Dentin Disordersmentioning

confidence: 99%

Dentin disorders: anomalies of dentin formation and structure

Coster¹

2009

Endodontic Topics

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Dentin defects represent a cluster of relatively rare heritable disorders affecting the formation and mineralization of dentin. Major clinical features include loss of enamel with extreme wear of exposed dentin, opalescent tooth discoloration, short roots with pulpal calcification and increased tooth mobility, and frequent periapical radiolucencies. Historically, these inherited conditions have been classified by Shields et al. (1) as either dentinogenesis imperfecta (DGI) types I, II and III, or dentin dysplasia (DD) types I and II. DGI type I is now universally designated as osteogenesis imperfecta with dentinogenesis imperfecta (OI/DGI) and is caused by type I collagen mutations. Among the genes expressing the non‐collagenous proteins in dentin, only the DSPP gene has been implicated in the etiology of DD type II and all DGI types, suggesting that heritable dentin defects can be viewed as a continuum rather than as distinct disease entities. Dental treatment strategies should aim to preserve the affected dentitions by protecting teeth from wear, restoring them to compliance with the specific structural characteristics of the defective dentin, and anticipating early tooth loss. Endodontic treatment of the affected teeth is very challenging because of the peculiar pulp morphology and most often has poor prognosis. If conventional therapy is not an option, periapical curettage and retrograde filling is a possible alternative, except in the case of teeth with short roots.

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Dentinogenesis imperfecta in a racial isolate with multiple hereditary defects

Cited by 37 publications

References 12 publications

Morphology of dental enamel and dentine–enamel junction in osteogenesis imperfecta

Morphology of dental enamel and dentine–enamel junction in osteogenesis imperfecta

Heritable dentin defects: Nosology, pathology, and treatment

Dentin disorders: anomalies of dentin formation and structure

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