Infant Odontogenic Myxoma: A specific entity

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Abstract

Introduction

In children, and specifically in infants, odontogenic myxomas are extremely rare. In infants, myxoma seems to display mostly the same clinical, radiological and pathological characteristics. This paper presents a series of odontogenic myxomas in infant patients.

Materials and methods

Four infant patients were included in this retrospective study. The clinical, radiological and pathological presentation was characterized and the treatment analysed.

Results

All patients presented with a rapidly evolving paranasal swelling. CT-scan showed a maxillary homogeneous unilocular and intraosseous tumour. In all cases, pathological examination revealed a loose myxoid stroma within stellate and spindle shaped cells. All patients underwent conservative surgery through a vestibular approach.

Conclusion

This patient series and a review of the literature demonstrates that odontogenic myxoma is specific in infant. We propose the name of Infant Odontogenic Myxoma for this entity.

Introduction

Myxoma is a rare benign mesenchymal tumour which can occur in many sites (Allen, 2000, Iatrou et al., 2012). In the jaw they are considered as odontogenic myxomas. Most odontogenic myxomas occur in the maxilla and mandible of adults in the third decade of life (Simon et al., 2004, Leiser et al., 2009). In children, and specifically in infants, odontogenic myxoma is uncommon, with only a few cases described in infants under the age of 2 years (Kansy et al., 2012). In the literature, myxoma in infants displays mostly the same clinical and radiological presentation (Kansy et al., 2012). This paper presents a series of odontogenic myxomas of infancy, treated in our department of paediatric maxillofacial surgery, and defines their clinical and radiological characteristics and optimal treatment.

Section snippets

Material and methods

Four cases of infants (under 2 years), with odontogenic myxoma treated and diagnosed in the Department of Maxillofacial Surgery from 2006 to 2012, and the records retrospectively examined. The following data were collected: patients' age, sex, medical history, clinical and photographic data, CT-scan analysis and histopathology report. The surgical management and recurrences were analysed.

Results (Table 1)

Four patients were included in our study, 3girls and 1 boy. The mean age was 19 months (range 14–23 months). None of the patients had personal or familial medical history. All patients were Caucasians. They all presented with a painless nasomaxillary swelling of 1–8 weeks (mean 3.75 weeks) duration. Two patients had had early recurrence after incomplete enucleation surgery. In one case, histopathology had concluded nodular fasciitis.

Physical examination revealed in all cases a non-tender, fixed

Discussion

This paper presents the largest series of odontogenic myxomas of infancy (children younger than 2 years). To our knowledge, 17 cases have been described in the international literature (Table 2).

Interestingly, all infants in our series and those previously described (20 overall), displayed the same clinical and radiological presentation (Tables 1 and 2).

In these 20 cases of Infant Odontogenic Myxoma, patients presented with a fixed and non-tender paranasal mass, potentially associated with

Conclusion

This series and previously published literature shows that odontogenic myxoma might be specific in infants. We propose the name of Infant Odontogenic Myxoma for this specific entity. In our definition, Infant Odontogenic Myxoma is a benign mesenchymal aggressive tumour, developing in an infant younger than 2 years. Clinically, it presents as a painless bony, paranasal swelling that may be associated with nasal or lacrimal duct obstruction. An intraosseous unilocular, lytic and homogeneous

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