Solitary Median Maxillary Central Incisor Syndrome: A Case Report
Suganya Balasubramanian
Keywords :
Case report, Incisor, Solitary median maxillary central incisor syndrome, Syndrome
Citation Information :
Balasubramanian S. Solitary Median Maxillary Central Incisor Syndrome: A Case Report. Int J Clin Pediatr Dent 2022; 15 (4):458-461.
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.
Scott DC. Absence of upper central incisor. Br Dent J 1958;104:247–248.
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
Hall RK. Solitary median maxillary central incisor (SMMCI) syndrome. Orphanet J Rare Dis 2006;1:12. DOI: 10.1186/1750-1172-1-12
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
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.
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
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
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
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
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
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
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
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
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
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
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
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
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
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
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
