Background:

Actinomycosis israeli is a gram-positive non-acid-fast bacterium and is the most common causative agent of actinomycosis. It usually presents as a brain abscess via a secondary spread from the cervicofacial region or lungs. However, presentation as an en-plaque lesion with bone involvement out of proportion to brain parenchyma is rare and needs to be acknowledged.
Case description:
We report a case of intracranial en-plaque lesion in a 47-year-old female with restriction of mouth opening and right third nerve palsy for 1 year. Imaging showed hyperostosis of the maxilla, temporal base, sphenoid, and zygoma with dural thickening suggestive of a meningioma. Surgical decompression yielded a granulomatous lesion along with empirical antitubercular therapy. However, patient succumbed to the infection and final histopathology revealed actinomycosis infection.
Conclusion:
Adequate knowledge along with clinical suspicion are the pillars to treating this unfortunate disease. Cheek swelling with bone involvement should be actively sought as risk factors. This case presents a unique opportunity to identify the fox among the sheep by elucidating the characteristic imaging findings and intraoperative view of this rare entity.