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Solitary median maxillary central incisor, short stature, choanal atresia/midnasal stenosis (SMMCI) syndrome.

Abstract

This article describes a series of 21 consecutive cases, each involving a solitary median maxillary central incisor; the patients were seen in the Department of Dentistry or the Victorian Clinical Genetics Unit, Murdoch Institute, at the Royal Children's Hospital, Melbourne, from 1966 to 1997. The spectrum of anomalies and associated features present in these cases--solitary median maxillary central incisor, choanal atresia, and holoprosencephaly--is described, and the literature related to the features, including genetic studies in these conditions, is reviewed. We relate our findings in these cases to current knowledge of developmental embryology. It is hoped that the findings, together with our interpretation of them, will help to clarify understanding of solitary median maxillary central incisor syndrome. This syndrome was previously considered a simple midline defect of the dental lamina, but it is now recognized as a possible predictor of holoprosencephalies of varying degrees in the proband, in members of the proband's family, and in the family's descendants.

Authors+Show Affiliations

Department of Dentistry, Royal Children's Hospital, Melbourne, Australia.No affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Journal Article
Research Support, Non-U.S. Gov't
Review

Language

eng

PubMed ID

9431535

Citation

Hall, R K., et al. "Solitary Median Maxillary Central Incisor, Short Stature, Choanal Atresia/midnasal Stenosis (SMMCI) Syndrome." Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontics, vol. 84, no. 6, 1997, pp. 651-62.
Hall RK, Bankier A, Aldred MJ, et al. Solitary median maxillary central incisor, short stature, choanal atresia/midnasal stenosis (SMMCI) syndrome. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1997;84(6):651-62.
Hall, R. K., Bankier, A., Aldred, M. J., Kan, K., Lucas, J. O., & Perks, A. G. (1997). Solitary median maxillary central incisor, short stature, choanal atresia/midnasal stenosis (SMMCI) syndrome. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontics, 84(6), pp. 651-62.
Hall RK, et al. Solitary Median Maxillary Central Incisor, Short Stature, Choanal Atresia/midnasal Stenosis (SMMCI) Syndrome. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1997;84(6):651-62. PubMed PMID: 9431535.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Solitary median maxillary central incisor, short stature, choanal atresia/midnasal stenosis (SMMCI) syndrome. AU - Hall,R K, AU - Bankier,A, AU - Aldred,M J, AU - Kan,K, AU - Lucas,J O, AU - Perks,A G, PY - 1998/2/12/pubmed PY - 1998/2/12/medline PY - 1998/2/12/entrez SP - 651 EP - 62 JF - Oral surgery, oral medicine, oral pathology, oral radiology, and endodontics JO - Oral Surg Oral Med Oral Pathol Oral Radiol Endod VL - 84 IS - 6 N2 - This article describes a series of 21 consecutive cases, each involving a solitary median maxillary central incisor; the patients were seen in the Department of Dentistry or the Victorian Clinical Genetics Unit, Murdoch Institute, at the Royal Children's Hospital, Melbourne, from 1966 to 1997. The spectrum of anomalies and associated features present in these cases--solitary median maxillary central incisor, choanal atresia, and holoprosencephaly--is described, and the literature related to the features, including genetic studies in these conditions, is reviewed. We relate our findings in these cases to current knowledge of developmental embryology. It is hoped that the findings, together with our interpretation of them, will help to clarify understanding of solitary median maxillary central incisor syndrome. This syndrome was previously considered a simple midline defect of the dental lamina, but it is now recognized as a possible predictor of holoprosencephalies of varying degrees in the proband, in members of the proband's family, and in the family's descendants. SN - 1079-2104 UR - https://www.unboundmedicine.com/medline/citation/9431535/Solitary_median_maxillary_central_incisor_short_stature_choanal_atresia/midnasal_stenosis__SMMCI__syndrome_ L2 - https://linkinghub.elsevier.com/retrieve/pii/S1079-2104(97)90368-1 DB - PRIME DP - Unbound Medicine ER -