Melkersson-Rosenthal syndrome is classically described as a triad of orofacial swelling, facial palsy, and fissured tongue. More often this syndrome presents in its oligosymptomatic forms. Melkersson-Rosenthal syndrome may not be as rare as suspected but rather a syndrome that often goes undiagnosed. Presented is the case of a mentally challenged man who was eventually diagnosed with Melkersson-Rosenthal syndrome only after being misdiagnosed and incorrectly treated for an odontogenic infection for more than a decade.