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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

Photomicrograph of bone biopsy of the buccal maxillary left canine region shows the characteristic features of segmental odontomaxillary dysplasia with thick trabeculae of viable woven bone, absence of cortical bone, and prominent irregular resting/reversal lines (hematoxylin and eosin stain). Original magnification × 100.
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f4A-imcrj-1-7: Photomicrograph of bone biopsy of the buccal maxillary left canine region shows the characteristic features of segmental odontomaxillary dysplasia with thick trabeculae of viable woven bone, absence of cortical bone, and prominent irregular resting/reversal lines (hematoxylin and eosin stain). Original magnification × 100.

Mentions: Bone biopsy after elevation of mucoperiosteal flap from the buccal maxillary left canine region was performed under general anesthesia. Histopathologic examination of the harvested bone sample revealed thick coarse bone trabculae of irregular and immature viable woven bone type with absence of lamellar bone and lacking osteoblastic/osteoclastic rimming. The bone marrow spaces were filled with active fine spindle cell stroma of fibrous connective tissue. Mitotic figures or pleomorphism were not observed. The histopathologic pattern was consistent with segmental odontomaxillary dysplasia (Figure 4).


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)

Photomicrograph of bone biopsy of the buccal maxillary left canine region shows the characteristic features of segmental odontomaxillary dysplasia with thick trabeculae of viable woven bone, absence of cortical bone, and prominent irregular resting/reversal lines (hematoxylin and eosin stain). Original magnification × 100.
© Copyright Policy
Related In: Results  -  Collection

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

f4A-imcrj-1-7: Photomicrograph of bone biopsy of the buccal maxillary left canine region shows the characteristic features of segmental odontomaxillary dysplasia with thick trabeculae of viable woven bone, absence of cortical bone, and prominent irregular resting/reversal lines (hematoxylin and eosin stain). Original magnification × 100.
Mentions: Bone biopsy after elevation of mucoperiosteal flap from the buccal maxillary left canine region was performed under general anesthesia. Histopathologic examination of the harvested bone sample revealed thick coarse bone trabculae of irregular and immature viable woven bone type with absence of lamellar bone and lacking osteoblastic/osteoclastic rimming. The bone marrow spaces were filled with active fine spindle cell stroma of fibrous connective tissue. Mitotic figures or pleomorphism were not observed. The histopathologic pattern was consistent with segmental odontomaxillary dysplasia (Figure 4).

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