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Enamel defects and Lyonization in focal dermal hypoplasia.

Abstract

We report a pattern of enamel hypoplasia in focal dermal hypoplasia similar to that found in females with X-linked amelogenesis imperfecta. Three cases of focal dermal hypoplasia are described, with specific focus on the oral and dental features. In these cases the teeth all had vertical grooving with notching of the incisal or cuspal tips. Also recorded were blunt roots of taurodont form with open apices and missing teeth in 1 case. Oral papillomas were present in 2 cases. The pattern of enamel defects is attributed to Lyonization, which is consistent with the pattern of skin and bone lesions typically seen in focal dermal hypoplasia. This supports the proposal that focal dermal hypoplasia is X-linked. The authors conclude that the pattern of dental defects in focal dermal hypoplasia is consistent with Lyonization.

Authors+Show Affiliations

Leeds Dental Institute, Leeds, United Kingdom. r.c.balmer@leeds.ac.ukNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

15583541

Citation

Balmer, Richard, et al. "Enamel Defects and Lyonization in Focal Dermal Hypoplasia." Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontics, vol. 98, no. 6, 2004, pp. 686-91.
Balmer R, Cameron AC, Adès L, et al. Enamel defects and Lyonization in focal dermal hypoplasia. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2004;98(6):686-91.
Balmer, R., Cameron, A. C., Adès, L., & Aldred, M. J. (2004). Enamel defects and Lyonization in focal dermal hypoplasia. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontics, 98(6), 686-91.
Balmer R, et al. Enamel Defects and Lyonization in Focal Dermal Hypoplasia. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2004;98(6):686-91. PubMed PMID: 15583541.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Enamel defects and Lyonization in focal dermal hypoplasia. AU - Balmer,Richard, AU - Cameron,Angus C, AU - Adès,Lesley, AU - Aldred,Michael J, PY - 2004/12/8/pubmed PY - 2005/2/3/medline PY - 2004/12/8/entrez SP - 686 EP - 91 JF - Oral surgery, oral medicine, oral pathology, oral radiology, and endodontics JO - Oral Surg Oral Med Oral Pathol Oral Radiol Endod VL - 98 IS - 6 N2 - We report a pattern of enamel hypoplasia in focal dermal hypoplasia similar to that found in females with X-linked amelogenesis imperfecta. Three cases of focal dermal hypoplasia are described, with specific focus on the oral and dental features. In these cases the teeth all had vertical grooving with notching of the incisal or cuspal tips. Also recorded were blunt roots of taurodont form with open apices and missing teeth in 1 case. Oral papillomas were present in 2 cases. The pattern of enamel defects is attributed to Lyonization, which is consistent with the pattern of skin and bone lesions typically seen in focal dermal hypoplasia. This supports the proposal that focal dermal hypoplasia is X-linked. The authors conclude that the pattern of dental defects in focal dermal hypoplasia is consistent with Lyonization. SN - 1079-2104 UR - https://www.unboundmedicine.com/medline/citation/15583541/Enamel_defects_and_Lyonization_in_focal_dermal_hypoplasia_ L2 - https://linkinghub.elsevier.com/retrieve/pii/S1079210404001623 DB - PRIME DP - Unbound Medicine ER -