Audrey Moreau1, Louise Galmiche2, Veronique Minard-Colin3, Martin Rachwalski4, Kahina Belhous5, Daniel Orbach6, Aline Joly1, Arnaud Picard7, Natacha Kadlub8. 1. Department of Maxillofacial and Plastic Surgery, Hôpital Universitaire Necker-Enfants Malades, 75015, Paris, France. 2. Department of Pathology, Hôpital Universitaire Necker-Enfants Malades, 75015, Paris, France; Université Paris Descartes, 75006, Paris, France. 3. Department of Pediatric and Adolescent Oncology, Institut Gustave Roussy, 94805, Villejuif, France. 4. Department of Maxillofacial and Plastic Surgery, Hôpital Universitaire Necker-Enfants Malades, 75015, Paris, France; Department of Pediatric Neurosurgery, National Reference Center for Craniofacial Malformations, Hôpital Universitaire Necker-Enfants Malades, 75015, Paris, France. 5. Department of Radiology, Hôpital Universitaire Necker-Enfants Malades, 75015, Paris, France. 6. Department of Pediatric Oncology, Institut Curie, 75248, Paris, France. 7. Department of Maxillofacial and Plastic Surgery, Hôpital Universitaire Necker-Enfants Malades, 75015, Paris, France; Université Paris Descartes, 75006, Paris, France; Laboratory for Oral and Molecular Pathology, INSERM, UMRS 1138, Centre de Recherche des Cordeliers, 75006, Paris, France. 8. Department of Maxillofacial and Plastic Surgery, Hôpital Universitaire Necker-Enfants Malades, 75015, Paris, France; Université Paris Descartes, 75006, Paris, France; Laboratory for Oral and Molecular Pathology, INSERM, UMRS 1138, Centre de Recherche des Cordeliers, 75006, Paris, France. Electronic address: natacha.kadlub@gmail.com.
Abstract
OBJECTIVES: Melanotic neuroectodermal tumor of infancy (MNTI) of the head and neck is a rare entity with uncertain clinical behavior. Radical surgical resection is the current recommended treatment, however this can cause severe aesthetic and functional sequelae. The aim of this study was to clinically characterize MNTIs and to stratify risk factors that may influence locoregional recurrence. METHODS: A retrospective multicenter study, including 11 patients from eight centers with a confirmed diagnosis of MNTI, was conducted. Epidemiological, clinical, radiological, pathological, and immunohistochemical examinations were reviewed. A statistical analysis using a t-test was conducted to calculate parameters correlating with tumor recurrence. RESULTS: MNTIs mainly occurred in the maxilla, with a mean age at diagnosis of 3.18 months (range: 0-6 months). Primary surgery was performed on 10 patients, with a clear margin resection on two patients. Overall recurrence rate was 27% with a survival of 100% at time of follow-up. No statistical correlation between recurrence rate, age at diagnosis, localization, resection margins, and pathological and immunohistochemical characteristics could be established. CONCLUSION: In our study, locoregional tumor recurrence did not seem to correlate with resection margins, so a conservative surgical approach may need to be considered to avoid functional and aesthetic sequelae.
OBJECTIVES:Melanotic neuroectodermal tumor of infancy (MNTI) of the head and neck is a rare entity with uncertain clinical behavior. Radical surgical resection is the current recommended treatment, however this can cause severe aesthetic and functional sequelae. The aim of this study was to clinically characterize MNTIs and to stratify risk factors that may influence locoregional recurrence. METHODS: A retrospective multicenter study, including 11 patients from eight centers with a confirmed diagnosis of MNTI, was conducted. Epidemiological, clinical, radiological, pathological, and immunohistochemical examinations were reviewed. A statistical analysis using a t-test was conducted to calculate parameters correlating with tumor recurrence. RESULTS: MNTIs mainly occurred in the maxilla, with a mean age at diagnosis of 3.18 months (range: 0-6 months). Primary surgery was performed on 10 patients, with a clear margin resection on two patients. Overall recurrence rate was 27% with a survival of 100% at time of follow-up. No statistical correlation between recurrence rate, age at diagnosis, localization, resection margins, and pathological and immunohistochemical characteristics could be established. CONCLUSION: In our study, locoregional tumor recurrence did not seem to correlate with resection margins, so a conservative surgical approach may need to be considered to avoid functional and aesthetic sequelae.
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