Background: In testicular germ cell tumors ( GCT), bone metastases are usually seen late in the disease progress and are almost always associated with involvement of other sites. However, isolated bone metastasis is an extremely rare finding in these patients. Case Report: A 43-year-old man was admitted to the neurosurgery department of our hospital suffering from dysarthria, ataxia, headaches and a progressive swelling above the parietooccipital region of the skull. Radiological, biochemical and pathologic tests showed that the lesion of the skull was an isolated skull metastasis as an initial manifestation of nonseminomatous GCT of the testis. Discussion: When a young patient presents with bone pain or painless swelling, even if it is an unusual site and isolated, testicular GCT should be considered as a differential diagnosis, as these lesions could be the first evidence of metastatic GCT.