Adult cases with isolated juvenile xanthogranuloma of the central nervous system are very rare. We report a case with dumbbell-type juvenile xanthogranuloma in the cervical spine. A 38-year-old man presented with moderate numbness of the right ring finger and right little finger and weakness of the right grip. Magnetic resonance imaging (MRI) showed an intra-spinal mass extending into the paravertebral area. The spinal cord was compressed by the lesion, which was isointense with the spinal cord on both T1- and T2-weighted imaging. Homogenous enhancement was observed after gadolinium administration. These findings favored a preoperative diagnosis of a rare tumor, rather than tumor of the nervous system. Complete surgical removal of the tumor was performed through hemilaminectomy combined with facetectomy between C7 and T1. Histological examination and immunohistochemical testing led to a diagnosis of juvenile xanthogranuloma. Two years after complete resection, MRI showed no recurrence. This appears to represent the first report of dumbbell-type juvenile xanthogranuloma in the cervical spine. Total removal of such lesions is recommended because of the high potential risk of tumor recurrence around the central nervous system.
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