We describe a patient who presented with a frontal sinus osteoma accompanied by a pneumocephalus causing aphasia. A -year old woman with a chief complaint of headache was referred to our outpatient ear, nose, and throat ENT clinic. A CT scan showed a large frontal sinus osteoma and pneumocephalus. Based on the standard language test for aphasia, she presented with aphasia including verbal paraphasia, and difficulty in word finding, reading, and auditory comprehension. She had no agrammatism, or apraxia of speech, and had no difficulties in word fluency or repetition. Kohs block-design test demonstrated that her IQ score was relatively low. After an endoscopic biopsy of her frontal sinus, a craniotomy with resection of the osteoma was performed. Her postoperative CT scan showed a remarkable reduction in the size of the pneumocephalus, and her linguistic and intelligence evaluation showed complete recovery. Her frontal sinus osteoma and linguistic and intelligent function have remained in the normal range for the last years. It is possible that transient aphasia due to pressure and ischemia might have occurred in the center of the orbital pars of the left inferior frontal gyrus, and in the networks between the cortical regions of language function. This is the first case report describing a frontal sinus osteoma accompanied by a pneumocephalus resulting in aphasia without other neurological symptoms.