Background: Mucoceles of paranasal sinuses are epithelial lined cysts that usually result from obstruction of osteo-meatal opening. They pose a diagnostic dilemma for clinicians due to their diverse symptoms mimicking either odontogenic or non-odontogenic lesions making them difficult to be diagnosed at an early stage. They affect the frontal (89%), ethmoid (8%), sphenoidal, and maxillary (2%) sinuses in adults. This case is unique in terms of the extent of the lesion and the rare involvement of the sphenoidal sinus. The current etiopathogenesis in the existing literature fails to explain this relatively rare involvement, which if not treated promptly can result in potentially fatal conditions such as cavernous sinus thrombosis, cranial nerve involvement, and acute pan sinusitis with bacteremia, brain abscess, and meningitis. Case Description: A 21-year-old Dravidian female patient presented with a right maxillary sinus mucocele on 5th May 2020. She had a history of swelling in the right cheek region for the past month, which had gradually progressed with no other associated symptoms. There was no relevant past medical history. Intraorally a firm swelling was present and the right upper first molar was grossly decayed, the second and third molars were Grade II mobile. Radiological evaluation of the lesion revealed an expansile cystic lesion occupying the entire maxillary sinus and obliteration of the sphenoid sinus. Surgical enucleation of the lesion with antrostomy was performed and the excisional biopsy report was suggestive of mucocele of the maxillary antrum. After a 3-year follow-up, the patient had no recurrence of the lesion and had an improved quality of life. Conclusions: Maxillary bone cysts present a diagnostic dilemma for clinicians mimicking an odontogenic lesion. The management of this lesion not only requires surgical enucleation but also nasal antrostomy, failing which recurrence may be expected or could result in fatal complications due to the spread and involvement of other adjacent vital structures, which could cause morbidity. Involvement of sphenoid sinus especially when infected could result in retrograde spread of infection leading to fatal complications. An accurate and timely understanding of this rare involvement can help clinicians tailor an appropriate treatment plan with minimal complications.