Unusual manifestations in X‐linked Amelogenesis Imperfecta

Lykogeorgos, T.; Duncan, Katherine R.; Crawford, P. J. M.; Aldred, MJ

doi:10.1046/j.1365-263x.2003.00484.x

Cited by 15 publications

(13 citation statements)

References 19 publications

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“…45-47 Cosegregation of hypoplastic enamel and delayed eruption in cases of AI suggests that the mutation causing this type of AI involves a gene expressed in ameloblasts as well as in surrounding odontogenic tissues. 47 The combination of defective enamel, dentin and eruption as seen in op/op mice has also been rarely reported in humans. 48 Thus, results in op/op models suggest that csCSF-1 may be a common link involved in mediating tooth matrix formation and eruption.…”

Section: Discussionmentioning

confidence: 99%

Targeted expression of csCSF-1 in op/op mice ameliorates tooth defects

Werner

Gluhak-Heinrich

Woodruff

et al. 2007

Archives of Oral Biology

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Objective-The aim of this study was to characterize the tooth phenotype of CSF-1-deficient op/ op mice and determine whether expression of csCSF-1 in these mice has a role in primary tooth matrix formation.Design-Ameloblasts and odontoblasts, isolated from wt/wt frozen sections using laser capture microdissection, were analyzed for csCSF-1, sCSF-1 and CSF-1R mRNA by RT-PCR. Mandibles, excised from 8 day op/op and wt/wt littermates, were examined for tooth morphology as well as amelogenin and DMP1 expression using in situ hybridization. Op/opCS transgenic mice, expressing csCSF-1 in teeth and bone using the osteocalcin promoter, were generated. Skeletal x-rays and histomorphometry were performed; teeth were analyzed for morphology and matrix proteins.Results-Normal dental cells in vivo express both CSF-1 isoforms and CSF-1R. Compared to wt/ wt, op/op teeth prior to eruption showed altered dental cell morphology and dramatic reduction in DMP1 transcripts. Op/opCS mice showed marked resolution of osteopetrosis, tooth eruption and teeth that resembled amelogenesis imperfecta-like phenotype. At 3 weeks, op/op teeth showed severe enamel and dentin defects and barely detectable amelogenin and DMP1. In op/opCS mice, DMP1 in odontoblasts increased to near normal and dentin morphology was restored; amelogenin also increased. Enamel integrity improved in op/opCS, although it was thinner than wt enamel.Conclusions-Results demonstrate that ameloblasts and odontoblasts are a source and potential target of CSF-1 isoforms in vivo. Expression of csCSF-1 within the tooth microenvironment is essential for normal tooth morphogenesis and may provide a mechanism for coordinating the process of tooth eruption with endogenous matrix formation.

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Section: Discussionmentioning

confidence: 99%

Targeted expression of csCSF-1 in op/op mice ameliorates tooth defects

Werner

Gluhak-Heinrich

Woodruff

et al. 2007

Archives of Oral Biology

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“…AI encompasses a diverse group of hereditary conditions that primarily affect the quality and/or the quantity of dental enamel. It has also been associated with non‐enamel anomalies, including taurodontism, congenitally missing teeth, dental follicle enlargement, delayed eruption, anterior open bite, crown resorption, and intrapulpal calcifications in unerupted and erupted teeth 8–12 . Review of the literature revealed reports of cases displaying several of these anomalies, but none associated so many defects or was as severe as the case described here.…”

Section: Discussionmentioning

confidence: 56%

A clinical and histological study of dental defects in a 10‐year‐old girl with pseudoxanthoma elasticum and amelogenesis imperfecta

Morrier

Roméas

Lacan

et al. 2008

Int J Paed Dentistry

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“…Clinically, skeletal anterior open bite is seen in approximately 50% of patients with AI of either X-linked or autosomal inheritance type. Non-enamel dental anomalies like taurodontism, elongation of pulp chamber due to apical displacement of root furcation and pulp calcifications occur with increased frequency in these patients when compared with their unaffected siblings (8). Increased incidence of pulp calcifications among AI patient could be due to local internal irritation of pulp due to loss of enamel and attrition (9).…”

Section: Discussionmentioning

confidence: 99%

Hypoplastic amelogenesis imperfecta with multiple impacted teeth - report of two cases

Reddy¹,

Nisha²,

Harish³

2010

J Clin Exp Dent

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Amelogenesis Imperfecta (AI) represents a group of developmental conditions, genomic in origin, which affect the structure and clinical appearance of enamel of all or nearly all the teeth in a more or less equal manner. It is usually inherited either as an X-linked, autosomal dominant or autosomal recessive trait. The enamel may be hypoplastic, hypomineralised or both and affected teeth may be discolored, sensitive or prone to disintegration. Diagnosis is based on the family history, pedigree plotting and meticulous clinical observation. The treatment of patients with AI should start with early diagnosis and intervention to prevent latter restorative problems. Herein, we present two case reports of hypoplastic amelogenesis imperfecta with oligodontia, multiple unerupted teeth, pulpal calcification, taurodontism and anterior deep bite who were provided with functional and esthetic rehabilitation. Key words: Amelogenesis imperfecta, multiple impacted teeth, oligodontia, taurodontism, pulpal calcification.Reddy SS, Aarthi Nisha V, Harish BN. Hypoplastic amelogenesis imperfecta with multiple impacted teeth -report of two cases. J Clin Exp Dent. 2010;2(4):e207-11.

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Unusual manifestations in X‐linked Amelogenesis Imperfecta

Cited by 15 publications

References 19 publications

Targeted expression of csCSF-1 in op/op mice ameliorates tooth defects

Targeted expression of csCSF-1 in op/op mice ameliorates tooth defects

A clinical and histological study of dental defects in a 10‐year‐old girl with pseudoxanthoma elasticum and amelogenesis imperfecta

Hypoplastic amelogenesis imperfecta with multiple impacted teeth - report of two cases

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