Intramedullary and Retroperitoneal Melanocytic Tumor Associated With Congenital Blue Nevus and Nevus Flammeus: An Uncommon Combination of Neurocutaneous Melanosis and Phacomatosis Pigmentovascularis—Case Report

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概要

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• Neurocutaneous melanosis (NCM) is a rare condition characterized by central nervous system melanocytic tumors associated with congenital melanocytic nevi. Phacomatosis pigmentovascularis (PPV) is an association of vascular nevus with pigmentary nevus. Aberrant maturation of neural crest-derived cells is considered to be related to pathogenesis in both conditions. However, association of NCM and PPV has not been reported to the best of our knowledge. Melanocytoma, which usually involves the leptomeninges or spinal cord, is extremely rare in the retroperitoneum. We present here a case of a patient with NCM, PPV, and melanocytic tumors in the spinal cord and retroperitoneum, which were treated surgically. A 40-year-old woman had a 2-year history of dysesthesia and weakness in the left leg. History included congenital giant blue nevus-like lesion in the trunk, a port-wine stain in the sacral area, and Caesarean section performed 8 years before, when diffuse pigmentation in the peritoneum was noted. Magnetic resonance (MR) imaging of the spine revealed an intramedullary tumor at T10 level with paramagnetic signal characteristics. The spinal cord tumor was totally removed, and the histological diagnosis was melanocytoma. Three months later, a left retroperitoneal mass with histological features of melanocytic tumor was removed. Neither tumors recurred and the patient stays ambulatory 4 years after the surgery. Multiple subtypes of melanocytic tumors with distinctive features of NCM and PPV can develop simultaneously, mimicking malignant melanoma. Gross total resection of each tumor, when indicated, is beneficial.

• 社団法人 日本脳神経外科学会の論文

著者

• Shimoda Mitsugi

  Second Department Of Surgery Dokkyo Medical University

• Kubota Keiichi

  Second Department Of Surgery

• Kurokawa Ryu

  Department Of Neurological Surgery Dokkyo University School Of Medicine

• Mori Shozo

  Second Department Of Surgery Tohoku University School Of Medicine

• Yamazaki Soji

  Department Of Dermatology Dokkyo Medical University

• Matsuda Hadzki

  Department Of Human Pathology Gunma University Graduate School Of Medicine

• Hatamochi Atsushi

  Department Of Dermatology Chiba University School Of Medicine

• HAYASHI Shujiro

  Department of Dermatology, Dokkyo Medical University, School of Medicine

• Kim Phyo

  Department Of Neurosurgery Dokkyo Medical University

• SHIMODA MITSUGI

  Second Department of Surgery, Dokkyo Medical University

• KAWAMOTO Toshiki

  Department of Neurologic Surgery, Dokkyo Medical University

• KIM Phyo

  Department of Neurologic Surgery, Dokkyo Medical University

• HAYASHI Shujiro

  Department of Dermatology, Dokkyo Medical University

• KUROKAWA Ryu

  Department of Neurologic Surgery, Dokkyo Medical University

• YAMAZAKI Soji

  Department of Dermatology, Dokkyo Medical University

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