International Journal of Clinical Pediatric Dentistry

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VOLUME 15 , ISSUE 4 ( July-August, 2022 ) > List of Articles

CASE REPORT

Solitary Median Maxillary Central Incisor Syndrome: A Case Report

Suganya Balasubramanian, Selvakumar Haridoss, Kavitha Swaminathan

Keywords : Case report, Incisor, Solitary median maxillary central incisor syndrome, Syndrome

Citation Information : Balasubramanian S, Haridoss S, Swaminathan K. Solitary Median Maxillary Central Incisor Syndrome: A Case Report. Int J Clin Pediatr Dent 2022; 15 (4):458-461.

DOI: 10.5005/jp-journals-10005-2418

License: CC BY-NC 4.0

Published Online: 17-12-2022

Copyright Statement:  Copyright © 2022; The Author(s).


Abstract

Aim: This report intends to present a case of solitary median maxillary central incisor syndrome (SMMCI) and its multidisciplinary team approach for diagnosis of other associated anomalies, with special emphasis on their management. Background: Solitary median maxillary central incisor syndrome is a unique developmental condition characterized by only maxillary central incisor and a series of developmental defects, appearing as a syndrome. The appearance of a single incisor may take place due to the union of two incisor teeth or the absence of tooth germs. The mechanism of the fusion is still uncertain. Case description: A 9-year-old female child reported with a chief complaint of pain in the right lower back tooth for the past 10 days. The presence of a single maxillary central incisor was an incidental finding. Then a detailed history and multidisciplinary evaluations revealed the diagnosis of SMMCI syndrome. Conclusion: The effort in diagnosing and managing this syndrome had a strong impact on the child's life of which the parent was highly motivated and got a better understanding of associated problems of overall development. Clinical significance: In SMMCI syndrome, the patient requires a multidisciplinary health team in order to improve their quality of life. It is of greater importance to diagnose the syndrome and treatment of these median line deformities.


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  1. Scott DC. Absence of upper central incisor. Br Dent J 1958;104:247–248.
  2. 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):651–662. DOI: 10.1016/s1079-2104(97)90368-1
  3. Hall RK. Solitary median maxillary central incisor (SMMCI) syndrome. Orphanet J Rare Dis 2006;1:12. DOI: 10.1186/1750-1172-1-12
  4. Kjaer I, Becktor KB, Lisson J, et al. Face, palate, and craniofacial morphology in patients with a solitary median maxillary central incisor. Eur J Orthod 2001;23(1):63–73. DOI: 10.1093/ejo/23.1.63
  5. Velasco DMM, Ramirez JAF, Nagano AY. Incisor syndrome single central midline fusion of the maxillary and mandibular permanent central incisors: report of clinical case. Rev Odont Mex 2005;9:37–41.
  6. Fleming P, Nelson J, Gorlin RJ. Single maxillary central incisor in association with mid-line anomalies. Br Dent J 1990;168(12):476–479. DOI: 10.1038/sj.bdj.4807238
  7. Harrison M, Calvert ML, Longhurst P. Solitary maxillary central incisor as a new finding in CHARGE association: a report of two cases. Int J Paediatr Dent 1997;7(3):185–189. DOI: 10.1046/j.1365-263x.1997.00236.x
  8. Arlis H, Ward RF. Congenital nasal pyriform aperture stenosis. Isolated abnormality vs developmental field defect. Arch Otolaryngol Head Neck Surg 1992;118(9):989–991. DOI: 10.1001/archotol.1992.01880090105027
  9. Wesley RK, Hoffman WH, Perrin J, et al. Solitary maxillary central incisor and normal stature. Oral Surg Oral Med Oral Pathol 1978;46(6):837–842. DOI: 10.1016/0030-4220(78)90318-3
  10. Buntinx I, Baraitser M. A single maxillary incisor as a manifestation of an ectodermal dysplasia. J Med Genet 1989;26(10):648–651. DOI: 10.1136/jmg.26.10.648
  11. Parentin F, Perissutti P. Solitary median maxillary central incisor, Duane retraction syndrome, growth hormone deficiency and duplicated thumb phalanx: a case report. Clin Dysmorphol 2003;12(2):141–142. DOI: 10.1097/00019605-200304000-00014
  12. Garcia de Paula e Silva FW, de Carvalho FK, Diaz-Serrano KV, et al. Solitary median maxillary central incisor in association with Goldenhar's syndrome: a case report. Spec Care Dentist 2007;27(3):105–107. DOI: 10.1111/j.1754-4505.2007.tb01749.x
  13. Lo FS, Lee YJ, Lin SP, et al. Solitary maxillary central incisor and congenital nasal pyriform aperture stenosis. Eur J Pediatr 1998;157(1):39–44. DOI: 10.1007/s004310050763
  14. Aughton DJ, AlSaadi AA, Transue DJ. Single maxillary central incisor in a girl with del (18p) syndrome. J Med Genet 1991;28(8):530–532. DOI: 10.1136/jmg.28.8.530
  15. Dolan LM, Willson K, Wilson WG. 18p-syndrome with a single central maxillary incisor. J Med Genet 1981;18(5):396–398. DOI: 10.1136/jmg.18.5.396
  16. Nanni L, Ming JE, Du Y, et al. SHH mutation is associated with solitary median maxillary central incisor: a study of 13 patients and review of the literature. Am J Med Genet 2001;102(1):1–10. DOI: 10.1002/1096-8628(20010722)102:1
  17. 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):417–423. DOI: 10.1007/s40368-013-0044-5
  18. Machado E, Machado P, Grehs B, et al. Solitary median maxillary central incisor syndrome: case report. Dental Press J Orthod 2010;15(4):55–61. DOI: 10.1590/S2176-94512010000400009
  19. Becktor KB, Sverrild L, Pallisgaard C, et al. Eruption of the central incisor, the intermaxillary suture, and maxillary growth in patients with a single median maxillary central incisor. Acta Odontol Scand 2001;59(6):361–366. DOI: 10.1080/000163501317153202
  20. Blackmore K, Wynne DM. A case of solitary median maxillary central incisor (SMMCI) syndrome with bilateral pyriform aperture stenosis and choanal atresia. Int J Pediatr Otorhinolaryngol 2010;74(8):967–969. DOI: 10.1016/j.ijporl.2010.05.018
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