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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

Frontal facial view of the patient with facial asymmetry and rough erythema involving left midface. Note hypopigmentation of upper lip and loss of vermilion border.
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f1-imcrj-1-7: Frontal facial view of the patient with facial asymmetry and rough erythema involving left midface. Note hypopigmentation of upper lip and loss of vermilion border.

Mentions: A 12-year-old Arab healthy male was referred by a general dental practitioner to the pediatric dental clinic with a concerned parent about unerupted teeth and facial swelling. His family and medical history were unremarkable except for a left facial eczema-like lesion which was treated repeatedly by a dermatologist for the past three years without significant improvement. Extraoral examination revealed mild left-sided facial swelling with hyperkeratotic diffuse erythematous lesion involving the left midface and extending from the left infraorbital margin to the commisure of the lip. Hypopigmentation of the left and middle parts of the upper lip was evident with unidentifiable vermilion border (Figure 1).


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)

Frontal facial view of the patient with facial asymmetry and rough erythema involving left midface. Note hypopigmentation of upper lip and loss of vermilion border.
© Copyright Policy
Related In: Results  -  Collection

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

f1-imcrj-1-7: Frontal facial view of the patient with facial asymmetry and rough erythema involving left midface. Note hypopigmentation of upper lip and loss of vermilion border.
Mentions: A 12-year-old Arab healthy male was referred by a general dental practitioner to the pediatric dental clinic with a concerned parent about unerupted teeth and facial swelling. His family and medical history were unremarkable except for a left facial eczema-like lesion which was treated repeatedly by a dermatologist for the past three years without significant improvement. Extraoral examination revealed mild left-sided facial swelling with hyperkeratotic diffuse erythematous lesion involving the left midface and extending from the left infraorbital margin to the commisure of the lip. Hypopigmentation of the left and middle parts of the upper lip was evident with unidentifiable vermilion border (Figure 1).

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