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Combined cutaneous findings with segmental odontomaxillary dysplasia: Review of the literature and proposal of a new clinical classification.

Yassin OM, Rihani FB - Int Med Case Rep J (2008)

Bottom Line: He presented with left midfacial diffuse hyperkeratotic erythematous lesion, ipsilateral hypopigmentation of upper lip with indistinct vermilion border, left-sided facial swelling, unilateral maxillary enlargement and ipsilateral failure of eruption of permanent teeth.Radiographic imaging showed abnormal bony trabeculation, impacted and missing teeth.Clinicians should be aware of its presence when encountering patients presenting with a facial cutaneous lesion especially when it is associated with facial asymmetry and unresponsive to treatment.

View Article: PubMed Central - PubMed

Affiliation: Consultant in pediatric dentistry, Chairman of Pediatric Dentistry Specialty at King Hussien Medical Center, Royal Medical Services, Amman, Jordan.

ABSTRACT
Segmental odontomaxillary dysplasia is characterized by variability of its clinical and radiological features, and may mimic other fibro-osseous lesions. We describe the case of a segmental odontomaxillary dysplasia in a 12-year-old male comprising of dermal, gingival, bony, and dental abnormalities. He presented with left midfacial diffuse hyperkeratotic erythematous lesion, ipsilateral hypopigmentation of upper lip with indistinct vermilion border, left-sided facial swelling, unilateral maxillary enlargement and ipsilateral failure of eruption of permanent teeth. Radiographic imaging showed abnormal bony trabeculation, impacted and missing teeth. Histopathologic findings showed characteristic features of segmental odontomaxillary dysplasia. We herein report a case of this rare unusual anomaly, review the literature, and propose a new clinical classification based on the limited number of previously reported cases in an attempt to categorize the clinical variants of the condition which might be helpful when treatment options are to be considered. Clinicians should be aware of its presence when encountering patients presenting with a facial cutaneous lesion especially when it is associated with facial asymmetry and unresponsive to treatment.

No MeSH data available.


Related in: MedlinePlus

Intraoral view of the maxillary arch showing unilateral left buccolingual gingivo-alveolar enlargement with missing canine and premolars.
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f2-imcrj-1-7: Intraoral view of the maxillary arch showing unilateral left buccolingual gingivo-alveolar enlargement with missing canine and premolars.

Mentions: Intraoral examination showed a dentition with all permanent teeth were present except the maxillary left permanent canine, and the maxillary left first and second premolars. The maxillary arch was asymmetric with enlarged alveolar bone buccolingually extending from the canine eminence to the maxillary tuberosity on the same side. The buccal gingival tissues covering the affected area was thickened and appeared fibrotic and partial obliteration of the labial sulcus was also noted (Figure 2).


Combined cutaneous findings with segmental odontomaxillary dysplasia: Review of the literature and proposal of a new clinical classification.

Yassin OM, Rihani FB - Int Med Case Rep J (2008)

Intraoral view of the maxillary arch showing unilateral left buccolingual gingivo-alveolar enlargement with missing canine and premolars.
© Copyright Policy
Related In: Results  -  Collection

Show All Figures
getmorefigures.php?uid=PMC3643048&req=5

f2-imcrj-1-7: Intraoral view of the maxillary arch showing unilateral left buccolingual gingivo-alveolar enlargement with missing canine and premolars.
Mentions: Intraoral examination showed a dentition with all permanent teeth were present except the maxillary left permanent canine, and the maxillary left first and second premolars. The maxillary arch was asymmetric with enlarged alveolar bone buccolingually extending from the canine eminence to the maxillary tuberosity on the same side. The buccal gingival tissues covering the affected area was thickened and appeared fibrotic and partial obliteration of the labial sulcus was also noted (Figure 2).

Bottom Line: He presented with left midfacial diffuse hyperkeratotic erythematous lesion, ipsilateral hypopigmentation of upper lip with indistinct vermilion border, left-sided facial swelling, unilateral maxillary enlargement and ipsilateral failure of eruption of permanent teeth.Radiographic imaging showed abnormal bony trabeculation, impacted and missing teeth.Clinicians should be aware of its presence when encountering patients presenting with a facial cutaneous lesion especially when it is associated with facial asymmetry and unresponsive to treatment.

View Article: PubMed Central - PubMed

Affiliation: Consultant in pediatric dentistry, Chairman of Pediatric Dentistry Specialty at King Hussien Medical Center, Royal Medical Services, Amman, Jordan.

ABSTRACT
Segmental odontomaxillary dysplasia is characterized by variability of its clinical and radiological features, and may mimic other fibro-osseous lesions. We describe the case of a segmental odontomaxillary dysplasia in a 12-year-old male comprising of dermal, gingival, bony, and dental abnormalities. He presented with left midfacial diffuse hyperkeratotic erythematous lesion, ipsilateral hypopigmentation of upper lip with indistinct vermilion border, left-sided facial swelling, unilateral maxillary enlargement and ipsilateral failure of eruption of permanent teeth. Radiographic imaging showed abnormal bony trabeculation, impacted and missing teeth. Histopathologic findings showed characteristic features of segmental odontomaxillary dysplasia. We herein report a case of this rare unusual anomaly, review the literature, and propose a new clinical classification based on the limited number of previously reported cases in an attempt to categorize the clinical variants of the condition which might be helpful when treatment options are to be considered. Clinicians should be aware of its presence when encountering patients presenting with a facial cutaneous lesion especially when it is associated with facial asymmetry and unresponsive to treatment.

No MeSH data available.


Related in: MedlinePlus