Summary Background Brachioradial pruritus is a rare form of pruritus localised to one or more brachioradial dermatomes, initially classified as a photodermatosis but which in fact is generally brought on by nervous compression. We report a case of a brachioradial pruritus revealing an intramedullary tumour. Patients and methods A 53-year-old man had presented pruritus for seven years under the left clavicle, then on the left forearm followed by the right forearm. Finally cervicodynia appeared associated with dysaesthesia of the two upper limbs, fulgurating pains and paresis of the left cubital region. The examination revealed suspended bilateral hypoaesthesia (C4, C5, C6, C7), proprioceptive disorders of the left upper limb, mild motor deficit in the left C8 area and tetrapyramidal syndrome. Cervical radiography did not show cervical osteoarthritis. The MRI revealed a bulky cervical intramedullary tumour extending from C2 to C6. After ruling out cavernoma by medullary angiography, surgery was performed and histopathological analysis of the complete lesion revealed a benign ependymoma. Four months later, this patient complained about residual pains requiring treatment with gabapentin and class 2 analgesics. Discussion The case presented underlines the possibility of a brachioradial pruritus revealing an intramedullary tumour. Ependymomas are usually seen in children and are frequently evoked in the presence of dysaesthesia. We report the third case of brachioradial pruritus revealing a medullary tumour. The two other cases involved syringomyelia revealed by pruritus in C5 and ependymoma revealed by pruritus in C5–C6. The patient with ependymoma had refused surgical treatment. Conclusion Atypical brachioradial pruritus complicated by neuropathic pains and disorders should prompt screening for a medullary tumour.