Structural deformities of deciduous teeth in patients with hypophosphatemic vitamin D-resistant rickets

Abe, Keiko; Ooshima, Takashi; Lily, Tong Sui Ming; Yasufuku, Y; Sobue, S

doi:10.1016/0030-4220(88)90165-x

Cited by 53 publications

(43 citation statements)

References 12 publications

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“…These defects create a way for microorganisms or toxic substances to the pulp, causing spontaneous necrosis and dental abscesses [11,18]. The present study showed that the anterior teeth of a 15-year-old XLH patient was affected, which is consistent with the previous study of McWhorter and Seale [19].…”

Section: Discussionsupporting

confidence: 91%

“…Radiographically, the enamel layer appears thinner while the dentin layer is more radiolucent. Some clinical studies reported the absence of enamel alterations in subjects affected by XLH [11]. However, Cremonesi et al, have shown by scanning electron microscopy, an irregular structured, slightly hypoplastic enamel in XLH teeth when compared to the images of control group [12].…”

Section: Introductionmentioning

confidence: 97%

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Endodontic Management of Patients With X Linked Hypophosphatemic Rickets: Case Series Report

Boukpessi¹,

Charreteur²,

Linglart³

et al. 2017

Dentistry

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Introduction: Familial hypophosphatemic rickets, the most common cause of inherited rickets, is in most cases transmitted as an X-linked dominant trait resulting from a mutation of the phosphate-regulating gene with homologies to endopeptidases on the X chromosome gene. This condition impairs bone, cement and dentin mineralization, resulting in skeletal and oral manifestations. Patients present spontaneous tooth abscesses that occur without any history of trauma or dental decay.

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Section: Discussionsupporting

confidence: 91%

Section: Introductionmentioning

confidence: 97%

Endodontic Management of Patients With X Linked Hypophosphatemic Rickets: Case Series Report

Boukpessi¹,

Charreteur²,

Linglart³

et al. 2017

Dentistry

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“…The Hyp mouse is a murine homologue of human X-linked hypophosphatemia (vitamin-D-resistant rickets). Histopathological studies of teeth of patients with X-linked hypophosphatemia have revealed hypomineralization of dentin indicated by abundant unmineralized interglobular dentin, broad predentin and irregular dentinal tubules [Abe et al, 1988]. Onishi et al [2005] proposed that Ocn is hyperexpressed in odontoblasts of the Hyp mouse, since Ocn mRNA reactivity by in situ hybridization was observed in odontoblasts alone.…”

Section: Discussionmentioning

confidence: 99%

Tributyltin Alters Osteocalcin, Matrix Metalloproteinase 20 and Dentin Sialophosphoprotein Gene Expression in Mineralizing Mouse Embryonic Tooth in vitro

Salmela

Alaluusua²,

Sahlberg³

et al. 2011

Cells Tissues Organs

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We showed in a previous in vitro study that tributyltin (TBT) arrests dentin mineralization and enamel formation in developing mouse tooth. The present aim was to investigate the effect of TBT on the expression of genes associated with mineralization of dental hard tissues. Embryonic day 18 mouse mandibular first molars were cultured for 3, 5 or 7 days and exposed to 1.0 µM TBT and studied by real-time quantitative polymerase chain reaction (RT-QPCR) for the expressions of osteocalcin (Ocn), alkaline phosphatase (Alpl), dentin matrix protein 1 (Dmp1), dentin sialophosphoprotein (Dspp) and matrix metalloproteinase 20 (Mmp-20).Ocn, Mmp-20 and Dspp, whose expressions showed changes in RT- QPCR, were further analyzed by in situ hybridization of tissue sections. In situ hybridization showed that TBT decreased Ocn expression in odontoblasts but increased the expression in the epithelial tooth compartment. In QPCR assays, the net effect in the whole tooth was increased expression. TBT also reduced Mmp-20 expression in ameloblasts and odontoblasts. Dspp expression varied but both QPCR assays and in situ hybridization showed a decreasing trend. TBT exposure had no clear effect on Alpl and Dmp1 expressions. Increased Ocn expression by epithelial enamel organ may inhibit dentin mineralization and enamel formation. Decreased Ocn, Mmp-20 and Dspp expressions in odontoblasts may indicate delayed cell differentiation, or TBT may specifically decrease the expression of genes involved in dentin mineralization. While decreased Mmp-20 expression by TBT in ameloblasts may impair enamel mineralization, the coincident reduction in Mmp-20 and Dspp expressions in odontoblasts may potentiate the delay of dentin mineralization.

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“…Sporadic cases are oft en initi ally detected by limb deformity or gait abnormality. The systemic fi ndings of XLH include bowed legs because of a body load showing immature skeletal bone calcifi cati on, spinal curvature deformiti es and beading of the ribs called rachiti c rosary [25][26][27] . Other causes of rickets include renal disease, medicati ons, and malabsorpti on syndromes (Table 1 8, [9][10][11][12][13][14][15] ).…”

Section: Types Of Ricketsmentioning

confidence: 99%

Types of Rickets, Dental and Histologic Findings: Review of the Literature

Tümen¹

2009

Pesq Bras Odontoped Clin Integr

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Introducti on: A disease that occurs during childhood, rickets is the failure of growing bone to mineralize. Many skeletal and radiographic changes can ocur because of the lack of calcifi ed osteoid and the buildup of unossifi ed carti lage. Proper bone formati on requires a complex interplay of several organs and chemicals, and vitamin D deserves special menti on because any disturbance in its producti on, absorpti on, or metabolism is paramount in the development of rickets. The pathophysiology of the disease is thought to be impaired phosphate transport, especially decreased phosphate resorpti on in the renal proximal tubule, as well as in the intesti ne. In most cases, the diagnosis is established with a thorough history and physical examinati on and confi rmed by laboratory evaluati on. Early diagnosis is essenti al because morbidity can be minimized if children are treated before eight months of age. Objecti ve: The aim of this literature review is to present various types of rickets with clinical features and the dental fi ndings, preventi ve measurements and treatments. Conclusion: The denti st as well as the pediatrician should be made aware of the features of this disorder so that early interventi on can prevent subsequent serious and more invasive dental procedures. KEYWORDS DESCRITORES Rickets;Hypophosphataemia; Oral manifestati ons; Therapeuti cs. RESUMO ABSTRACTIntrodução: O raquiti smo, uma doença que ocorre durante a infância, é a falta de crescimento ósseo necessário para a mineralização. Diversas alterações radiográfi cas esqueléti cas podem ocorrer devido a ausência de osteóide calcifi cado e a formação de carti lagem. A formação óssea adequada requer uma complexa interação de vários órgãos e produtos químicos, e a vitamina D merece uma menção especial, pois qualquer alteração na sua produção, absorção ou no seu metabolismo é fundamental para o desenvolvimento do raquiti smo. Acredita-se que a fi siopatologia da doença está no transporte inadequado do fosfato, especialmente na diminuição da reabsorção de fosfato no túbulo renal proximal bem como no intesti no. Na maioria dos casos, o diagnósti co é estabelecido com uma história completa e exame fí sico, e confi rmado por exames laboratoriais. O diagnósti co precoce é essencial, pois a morbidade pode ser minimizada se as crianças forem tratadas antes dos oito meses de idade. Objeti vo: Apresentar os vários ti pos de raquiti smo com suas característi cas clínicas, aspectos dentários, medidas preventi vas e tratamento. Conclusão: O cirurgião-denti sta, bem como o pediatra, devem ser informados das característi cas desta desordem para que a intervenção precoce possa evitar seqüelas subsequentes e procedimentos odontológicos mais invasivos.

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Structural deformities of deciduous teeth in patients with hypophosphatemic vitamin D-resistant rickets

Cited by 53 publications

References 12 publications

Endodontic Management of Patients With X Linked Hypophosphatemic Rickets: Case Series Report

Endodontic Management of Patients With X Linked Hypophosphatemic Rickets: Case Series Report

Tributyltin Alters Osteocalcin, Matrix Metalloproteinase 20 and Dentin Sialophosphoprotein Gene Expression in Mineralizing Mouse Embryonic Tooth in vitro

Types of Rickets, Dental and Histologic Findings: Review of the Literature

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