European Archives of Paediatric Dentistry

, Volume 9, Issue 4, pp 245–248 | Cite as

Segmental odontomaxillary dysplasia — a rare disorder

  • S. K. BhatiaEmail author
  • N. Drage
  • A. J. Cronin
  • M. L. Hunter
Case Report


Background: Segmental odontomaxillary dysplasia (SOD) is a rare developmental disorder of the maxilla characterised by abnormal growth and maturation of bone, teeth and gingiva of the affected segment. Due to the rarity of the disorder, the full range of clinical manifestations is unknown. Since there are concerns that cases may be subject to misdiagnosis, this report aims to increase awareness of the condition among paediatric dentists. Case Report: A 7-year-old boy was initially referred regarding failure of eruption of maxillary primary molars. Characteristic clinical and radiographic findings led to the diagnosis of SOD. Interestingly, the patient presented with unilateral ectopic eyelashes, a finding previously unreported in association with this condition. Treatment: Reassurance was provided regarding the benign nature of the condition. While no active intervention was needed at the time of diagnosis, close monitoring is essential. Future management may require surgical, orthodontic and prosthetic input. Follow-up: A period of four years elapsed between initial presentation of the condition and its definitive diagnosis. No significant increase in the unilateral maxillary swelling was noted. The patient will continue to be monitored on a regular basis. Conclusion: SOD is a rare cause of facial asymmetry. Prompt diagnosis can reassure both patient and healthcare professionals. Reporting of cases is encouraged to help determine the full range of clinical manifestations and establish a management protocol.

Key words

segmental odontomaxillary dysplasia maxillary swelling vertical bony trabeculae ectopic cilia 


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  1. Danforth RA, Raymond JM, Abrams AM, Handlers JP. Segmental Odontomaxillary Dysplasia. Report of eight cases and comparison with hemimaxillofacial dysplasia. Oral Surg, Oral Med, Oral Pathol 1990; 770: 81–5CrossRefGoogle Scholar
  2. DeSalvo MS, Copete MA, Riesenberfer RE, Cleveland DB, Chen S. Segmental Odontomaxillary dysplasia (hemimaxillofacial dysplasia): Case report. Paed Dent 1996; 18(2): 154–156Google Scholar
  3. Drake DL. Segmental odontomaxillary dysplasia: An unusual orthodontic challenge. Am J Orthod Dentofacial Orthop 2003; 123: 84–86PubMedCrossRefGoogle Scholar
  4. Duker-Elder S. Ed. System of Ophthamology Vol III: Normal and abnormal development Pt 2: Congenital deformities. St Louise: CV Mosby, 1964:872–881Google Scholar
  5. Johnson R. An unusual case of an extra row of eyelashes in the superior eyelid skin crease. Clin Exp Optom 2006; 89:5: 322–324Google Scholar
  6. Jones CJ, Ford JF. Simultaneous occurrence of segmental odontomaxillary dysplasia and Becker’s nevus. J Oral Maxillofac Surg 1999; 57:1251–1254PubMedCrossRefGoogle Scholar
  7. Miles DA, Lovas JL, Cohen MM. Hemimaxillofacial dysplasia: A newly recognised disorder of facial asymmetry, hypertricosis of the facial skin, unilateral enlargement of the maxilla, and hypoplastic teeth in two patients. Oral Surg Oral Med, Oral Pathol 1987; 64: 445–448CrossRefGoogle Scholar
  8. Packota GV, Pharoah MJ, Petrikowski CG. Radiographic features of segmental odontomaxillary dysplasia: a study of 12 cases. Oral Surg, Oral Med, Oral Pathol, Oral Radiol, Endod 1996; 82(5):577–584CrossRefGoogle Scholar
  9. Paticoff K, Marion RW, Shprintzen RJ, Shanske AL, Eisig SB. Hemimaxillofacial dysplasia. A report of two new cases and furthur delineation of the disorder. Oral Surg, Oral Med, Oral Pathol, Oral Radiol, Endod 1997; 83: 484–488CrossRefGoogle Scholar
  10. Prusack N, Pringle G, Scotti V, Chen S. Segmental Odontomaxillary Dysplasia. A case report and review of literature. Oral Surg, Oral Med, Oral Pathol Oral Radiol Endod 2000; 90: 483–488CrossRefGoogle Scholar

Copyright information

© Adis International 2008

Authors and Affiliations

  • S. K. Bhatia
    • 1
    Email author
  • N. Drage
    • 2
  • A. J. Cronin
    • 3
  • M. L. Hunter
    • 4
  1. 1.Paediatric DentistryUniversity Dental HospitalCardiff, WalesUK
  2. 2.Oral and Maxillofacial RadiologyUniversity Dental HospitalCardiff, WalesUK
  3. 3.Oral and Maxillofacial SurgeryUniversity Dental HospitalCardiff, WalesUK
  4. 4.School of DentistryCardiff UniversityCardiff, WalesUK

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