Introduction: Ceruminous adenomas are benign neoplasms arising from ceruminous glands. These tumours should be limited to the external ear due to the location of these glands. Ceruminous adenomas occurring within the middle ear are exceedingly rare, with <10 well-documented cases reported in the English literature. We present the case of a histopathologically confirmed ceruminous adenoma of the middle ear in a 44-year-old male, initially suspected to be a cholesteatoma. Case: A 44-year-old male was referred to otolaryngology with a chronic left tympanic membrane perforation and concern for cholesteatoma. His medical history included type II diabetes mellitus, former smoking, and had multiple ventilation tube insertions during childhood with longstanding with bilateral chronic otorrhoea. Otoscopic examination demonstrated a normal external auditory canal with a large subtotal tympanic membrane perforation and keratinisation in the attic, raising suspicion for a cholesteatoma. Preoperative CT petrous temporal bones and MRI acoustic series also suggested cholesteatoma. A left cortical tympanomastoidectomy was performed. Intraoperatively, findings were inconsistent with cholesteatoma, instead revealing a 3mm tegman antri dehiscence with a CSF leak and extensive granulation tissue over the dura, throughout the mastoid cavity, and middle ear including the eustachian tube opening. Histopathological analysis confirmed a ceruminous adenoma of the middle ear. Subsequent imaging demonstrates similar appearances on the contralateral side. Discussion: Ceruminous adenomas are rare, with involvement of the middle ear being exceptionally uncommon. This case resulted in an unexpected intraoperative finding despite clinical and radiological features suggesting cholesteatoma. Therefore, although uncommon, this case highlights the importance of considering ceruminous adenoma within the middle ear as a possible differential when evaluating a soft tissue middle ear mass of unknown origin.