Infant Odontogenic Myxoma: Case Report and Literature Review of a Specific Entity Recently Described

Abstract

Odontogenic myxomas are benign mesenchymal neoplasm most of them diagnosed in adults. They are uncommon in the paediatric population and exceptional in infants, with only 21 cases reported in the literature under the age of 2 years. We present a new case of infant odontogenic myxoma, that share the same clinical and radiological presentation with the cases described in the literature. They all presented with a painless paranasal swelling of short-term evolution, usually a few weeks duration (while in children or adults tumours usually develop slowly) and a well-defined, intraosseous, expansible lytic tumour of 3 cm average size in CT-scan examination. Most cases underwent enucleation and curettage with a very low rate of recurrences (4.76%). The aim of this article is to report a new case of this exceptional tumour, whose diagnosis was established at histologic examination. We focus on the importance to undergo a conservative approach in this infant population to minimize the surgical morbidity.These tumours do not metastasize and although malignant transformation to myxosarcoma has been reported is a rare event [3]. Case ReportA healthy 21-month-old male, with no relevant personal or family medical history, presented to our hospital with a persistent swelling on the right side of his face after a minor trauma a few weeks before. The patient had persistent swelling that had no regressed. On physical examination there was an indurated, fixed 4cm mass in his right nasolabial groove, adjacent to the right anterior maxillary wall. The lesion did not enlarge with crying. The overlying skin was normal. Eye position and extra ocular motion were normal. An intraoral examination showed obliteration of the maxillary vestibule. Clinically the lesion looked like a mucocele.In CT-scan examination there was a low-density lesion arising within the anterior medial aspect of the left maxillary bone with erosion of the maxillary sinus and the lateral nasal wall (Figure 1). There was separation of the mass from the nasal-lacrimal duct. Taking into account the history of trauma to the area, the CT scan concluded that the lesion was compatible with post-traumatic cyst.Enucleation of the tumour was performed with curettage of the surrounding bone through a vestibular incision. The patient recovered uneventfully from surgery.On gross pathology, the largest dimension of the lesion was 4cm and it was gelatinous with pale brown colour. Histology revealed a myxoid tumour with haphazardly arranged stellate to spindle-shaped cells in a mucoid-rich intercellular matrix (Figure 2). The cells had an eosinophilic cytoplasm, small round hyperchromatic nuclei and fine chromatin. There was not cellular pleomorphism or nuclear atypia. Immunohistochemical staining was positive for vimentin and

Odontogenic myxoma in an 8-year-old girl: A case report with review of literature

“…stated that conservative surgical excision done in infants did not show any clinical or radiographic evidence of recurrence for 3-year posttreatment follow-up. [ 17 ] Recurrence is minimized with extensive partial or total resection of the lesion. While doing resection particularly in the maxilla, proximity of vital structures should be considered.…”

Pediatric Odontogenic Myxoma: A Case Report and a Systematic Review of the Literature

“…25 For tumor removal, surgeons are faced with the dilemma of an unencapsulated, locally aggressive, benign tumor that has reported high recurrence rates with conservative enucleation in the adult population (25%-45%). 9,10,40 Recurrence of OM has been related to the incomplete removal of nests and loculations or pockets of the myxoid tumor beyond clinical or radiologically margins. 44 This has led to the recommendations of resection with margins of 10 to 15 mm to reduce the rates of recurrence.…”