A 43 year old male presented with complaints of neck pain radiating to bilateral upper limb with weakness of bilateral upper limbs. Examination revealed a loss of cervical lordosis with restricted range of motion of the cervical spine .Neurological examination revealed a weakness of finger abduction and adduction. Blood investigations showed an elevated erythrocyte sedimentation rate, while the rest were within normal limits.MRI scan of the cervical spine showed an hyperintense lesion of the first thoracic vertebral body. CT guided biopsy was attempted followed by open biopsy of the lesion and specimen was sent for histopathological examination which was later reported as Langerhans cell histiocytosis and the patient was treated with chemotherapy for the same. Despite the rarity of the condition, the possibility of LCH should be taken into account in these circumstances. This case shows the role of chemotherapy in management and draws attention towards early detection in order to prevent neurological complications with respect to the rarity of the disease and the site of the bone involvement.

Keywords : Langerhans Cell Histiocytosis, Chemotherapy.