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Solitary median maxillary central incisor syndrome (SMMCI) with congenital nasal puriform aperture stenosis: literature review and case report with comprehensive dental treatment and 14 years follow-up.
Eur Arch Paediatr Dent 2013; 14(6):417-23EA

Abstract

BACKGROUND

Solitary median maxillary central incisor syndrome [SMMCI] is an extremely rare anomaly, especially when no other abnormalities are present. The defect is often found together with various nasal abnormalities and short stature with or without decreased levels of growth hormone. In more severe cases, SMMCI has been associated with holoprosencephaly, the CHARGE and the VACTERL association. Also, published sporadic cases have been related with rare variants of ectodermal dysplasia, chromosomal abnormalities, precocious puberty, hypothalamic hamartoma, congenital heart defects, physical/mental retardation, genital hypoplasia and ear abnormalities. For these reasons when the initial diagnosis is made by the paediatric dentist, ENT, neurological and paediatric evaluations are essential.

CASE REPORT

A 4-year-old boy with SMMCI was referred for dental treatment. Clinical/radiographic examination revealed a symmetrical primary and permanent SMMCI, a skeletal Class I and a unilateral crossbite. Medical history indicated respiratory distress and surgery soon after birth due to congenital nasal puriform aperture stenosis. Gradual orthodontic treatment started at the age of 4 years and completed at the age of 13 years. Following maxillary expansion, upper lateral segments were moved backwards and anterior space was created for accommodating a second central incisor. Retainers with a supplementary acrylic incisor were provided for aesthetic and functional replacement until the age of 16 years, when a fixed Maryland ceramic bridge was placed.

FOLLOW-UP

Two years recall, at the age of 18 years, revealed a satisfactory and stable aesthetic and functional result.

CONCLUSION

Successful dental management of SMMCI patients is possible, following a detailed long-lasting treatment plan requiring multidisciplinary paediatric dental, orthodontic and prosthetic approach.

Authors+Show Affiliations

Private Paediatric Dental Practice, 2 Papadiamantopoulou Street, 115 28, Athens, Greece.No affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Case Reports
Journal Article
Review

Language

eng

PubMed ID

23775592

Citation

Lygidakis, N N., et al. "Solitary Median Maxillary Central Incisor Syndrome (SMMCI) With Congenital Nasal Puriform Aperture Stenosis: Literature Review and Case Report With Comprehensive Dental Treatment and 14 Years Follow-up." European Archives of Paediatric Dentistry : Official Journal of the European Academy of Paediatric Dentistry, vol. 14, no. 6, 2013, pp. 417-23.
Lygidakis NN, Chatzidimitriou K, Petrou N, et al. Solitary median maxillary central incisor syndrome (SMMCI) with congenital nasal puriform aperture stenosis: literature review and case report with comprehensive dental treatment and 14 years follow-up. Eur Arch Paediatr Dent. 2013;14(6):417-23.
Lygidakis, N. N., Chatzidimitriou, K., Petrou, N., & Lygidakis, N. A. (2013). Solitary median maxillary central incisor syndrome (SMMCI) with congenital nasal puriform aperture stenosis: literature review and case report with comprehensive dental treatment and 14 years follow-up. European Archives of Paediatric Dentistry : Official Journal of the European Academy of Paediatric Dentistry, 14(6), pp. 417-23. doi:10.1007/s40368-013-0044-5.
Lygidakis NN, et al. Solitary Median Maxillary Central Incisor Syndrome (SMMCI) With Congenital Nasal Puriform Aperture Stenosis: Literature Review and Case Report With Comprehensive Dental Treatment and 14 Years Follow-up. Eur Arch Paediatr Dent. 2013;14(6):417-23. PubMed PMID: 23775592.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Solitary median maxillary central incisor syndrome (SMMCI) with congenital nasal puriform aperture stenosis: literature review and case report with comprehensive dental treatment and 14 years follow-up. AU - Lygidakis,N N, AU - Chatzidimitriou,K, AU - Petrou,N, AU - Lygidakis,N A, Y1 - 2013/06/18/ PY - 2013/04/08/received PY - 2013/04/30/accepted PY - 2013/6/19/entrez PY - 2013/6/19/pubmed PY - 2016/4/24/medline SP - 417 EP - 23 JF - European archives of paediatric dentistry : official journal of the European Academy of Paediatric Dentistry JO - Eur Arch Paediatr Dent VL - 14 IS - 6 N2 - BACKGROUND: Solitary median maxillary central incisor syndrome [SMMCI] is an extremely rare anomaly, especially when no other abnormalities are present. The defect is often found together with various nasal abnormalities and short stature with or without decreased levels of growth hormone. In more severe cases, SMMCI has been associated with holoprosencephaly, the CHARGE and the VACTERL association. Also, published sporadic cases have been related with rare variants of ectodermal dysplasia, chromosomal abnormalities, precocious puberty, hypothalamic hamartoma, congenital heart defects, physical/mental retardation, genital hypoplasia and ear abnormalities. For these reasons when the initial diagnosis is made by the paediatric dentist, ENT, neurological and paediatric evaluations are essential. CASE REPORT: A 4-year-old boy with SMMCI was referred for dental treatment. Clinical/radiographic examination revealed a symmetrical primary and permanent SMMCI, a skeletal Class I and a unilateral crossbite. Medical history indicated respiratory distress and surgery soon after birth due to congenital nasal puriform aperture stenosis. Gradual orthodontic treatment started at the age of 4 years and completed at the age of 13 years. Following maxillary expansion, upper lateral segments were moved backwards and anterior space was created for accommodating a second central incisor. Retainers with a supplementary acrylic incisor were provided for aesthetic and functional replacement until the age of 16 years, when a fixed Maryland ceramic bridge was placed. FOLLOW-UP: Two years recall, at the age of 18 years, revealed a satisfactory and stable aesthetic and functional result. CONCLUSION: Successful dental management of SMMCI patients is possible, following a detailed long-lasting treatment plan requiring multidisciplinary paediatric dental, orthodontic and prosthetic approach. SN - 1818-6300 UR - https://www.unboundmedicine.com/medline/citation/23775592/Solitary_median_maxillary_central_incisor_syndrome__SMMCI__with_congenital_nasal_puriform_aperture_stenosis:_literature_review_and_case_report_with_comprehensive_dental_treatment_and_14_years_follow_up_ L2 - https://dx.doi.org/10.1007/s40368-013-0044-5 DB - PRIME DP - Unbound Medicine ER -