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Intracranial intermediate-grade meningeal melanocytoma with increased cellular proliferative index: an illustrative case associated with a nevus of Ota

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Abstract

Solitary primary melanocytic tumors of the central nervous system (CNS) represent a spectrum of lesions ranging from well-differentiated melanocytoma to melanoma. The association of a meningeal melanocytoma with an ipsilateral nevus of Ota is extremely rare, with only six cases reported in the literature to date. Only a minority of melanocytic tumors correspond to lesions of intermediate-grade malignancy, whose biological behavior and outcome remain undetermined. We report a new case of a 25-year-old man with a giant fronto-temporal intracranial meningeal melanocytoma associated with a congenital nevus of Ota, who suffered an acute neurological deterioration requiring an emergent surgical procedure. Despite total removal of the lesion, the patient did not recover from the operation due to a malignant infarct of the right hemisphere. The lesion showed no histological criteria of malignancy but did exhibit an elevated (8%) MIB-1/Ki-67 cell proliferative index, which suggested an intermediate-grade melanocytoma. In addition, its metabolic profile, determined by in vivo proton magnetic resonance spectroscopy (MRS), was similar to that observed in most high-grade gliomas. To our knowledge, this is the first case of a meningeal melanocytoma of intermediate grade associated with a nevus of Ota reported in the literature. Only two additional intermediate-grade melanocytomas showing an elevated MIB-1/Ki-67 labeling index have been previously reported. The MIB-1/Ki-67 labeling index may have potential prognostic value in helping the clinician to predict an aggressive clinical behavior and/or malignant progression for primary melanocytic neoplasms of the CNS.

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Acknowledgements

We thank Drs. Motoo Nagane, Marie-Dominique Piercecchi, and Vafa Rahimi-Movaghar for providing us some lacking information from their publications that was finally included in Table 1. We also thank Drs. Roser, Wang, and Rades for their kind responses to our requests. Finally we deeply appreciate helpful assistance by Dr. Mostafa El-Khashab who kindly submitted his recently published paper included in the reviewed version of our manuscript.

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Authors and Affiliations

  1. Department of Neurosurgery, La Princesa University Hospital, C/Diego de León 62, 28006, Madrid, Spain

    Marta Navas, José M. Pascual, Manuel Pedrosa, Rodrigo Carrasco, Pedro Martínez & Rafael García de Sola

  2. Department of Pathology, La Princesa University Hospital, Madrid, Spain

    Javier Fraga

  3. Department of Neuroradiology, La Princesa University Hospital, Madrid, Spain

    Rafael Manzanares

  4. Johns Hopkins University School of Medicine, Baltimore, MD, USA

    Sophia Shakur

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  1. Marta Navas
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Correspondence to Marta Navas.

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Navas, M., Pascual, J.M., Fraga, J. et al. Intracranial intermediate-grade meningeal melanocytoma with increased cellular proliferative index: an illustrative case associated with a nevus of Ota. J Neurooncol 95, 105–115 (2009). https://doi.org/10.1007/s11060-009-9907-3

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