Congenital defect to the foramen of Huschke with a manifestation as temporomandibular joint (TMJ) soft tissue herniation in a radiological study is rare. The patient may present with nonspecific symptoms such as otalgia, mandibular joint pain, tinnitus and conductive hearing loss, and scarcely cervicofacial subcutaneous emphysema. Here, we report a patient presented with cervicofacial subcutaneous emphysema secondary to a congenital defect of the foramen of Huschke. A 45-year-old gentleman presented with right-sided neck swelling and right otalgia with a crackling sound over the right ear upon chewing. Examination shows right-sided fullness with subcutaneous emphysema from the zygoma to the upper neck. Otherwise, it is non-tender with no skin changes, and the facial nerve is intact. Otoendoscopy shows erythematous soft tissue bulging of the anterior wall of the right external auditory canal (EAC) upon closing the mouth and prolapsing upon mouth opening. The right tympanic membrane was intact. Contrast-enhanced computer tomography (CECT) of the neck and temporal region revealed extensive cervicofacial subcutaneous emphysema with a bony defect at the anterior wall of the right EAC, indicating fistulous communication between the right EAC and TMJ. The subcutaneous emphysema resolved on the treatment of the right otitis externa. The patient is subjected to a combined approach of open and endoscopic-assisted repair of the anterior EAC wall defect. TMJ herniation into the anterior EAC is rare; however, the patient presentation may vary. CECT is the gold standard for diagnosing and facilitating treatment options. Treatment choice is based on the patient's condition, including conservative or surgical intervention.
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