A 60-year-old male presented to the ENT OPD of Assam Medical College Dibrugarh, with purulent foul smelling discharge and severe pain in the right ear associated with marked hearing loss. There was no history of tinnitus and vertigo. The patient gives history of similar symptoms of pain and purulent discharge from the right ear for which he underwent ear surgery twice about 40 years back, but no documentation regarding the procedure was available with the patient.On otoscopic examination central perforation of right tympanic membrane was noted while left tympanic membrane was intact. Pure tone audiometry showed hearing loss of 60 decibel. On nasal examination there was no evidence of nasal papilloma. CT scan showed right sided chronic supporative otitis media of atticoantral type.Based on his chief complaints, the patient was considered for radical mastoidectomy. But immediately after 2 months of surgery, he again presented with similar symptoms of severe pain and purulent fowl smelling discharge from the right ear along with gaping in the right post auricular wound with exposed mastoid cavity filled with purulent discharge.Repeat CT scan showed postoperative bony defect in right petromastoid region with extensive soft tissue lesion with erosive bone changes on right side [Table/ Fig-1a-d].The patient was again planned for mastoid exploration. Extensive granulation tissues were removed following mastoidectomy. The histopathological examination of the tissue removed revealed picture of inverted papilloma with marked dysplasia [Table/ Fig-2]. Immunohistochemistry was done to tag mitosis by proliferative marker Ki67 [16/10 HPF] showed moderate degree of proliferative activity.The patient was then treated with postoperative external beam radiation in a cobalt-60 teletherapy machine (Theratron 780 C). A total dose of 54 Gy was delivered in 30 daily fractions of 180 cGy with anterior and posterior oblique wedge fields. After the completion of the course of radiotherapy, there was no pain and discharge from the right ear.Patient was followed up at one month and then every three month. MRI of the temporomastoid region was done in each follow-up using Siemens Avanto 1.5 Tesla machine.There were no symptoms of pain and ear discharge at the end of one month except having mixed hearing loss. No signs of Inverted papilloma or Schneiderian papilloma arising primarily from middle ear and mastoid is a very rare entity. It is a benign and locally aggressive condition, with increased risk of recurrence and malignant transformation. Surgery is the primary treatment. Radiation therapy should be considered in case of malignant transformation, recurrent or inoperable disease. Here we are reporting a case of recurrent inverted papilloma with dysplastic changes arising from mastoid cavity in a 60-year-old male. The patient was treated with surgery followed by radiotherapy.