Olmsted syndrome with oral involvement, including premature teeth loss

Olmsted syndrome is a rare palmoplantar keratodermal disease that has not previously been reported to have an association with periodontal disease. The aim of this study is to report and document a case of Olmsted syndrome with evidence of severe periodontal disease.

A 38-year old Saudi male patient presented to the dental clinic diagnosed previously with Olmsted syndrome. Clinical and radiographic examinations were done and provided evidence of the typical clinical findings in Olmsted syndrome and evidence of severe periodontal disease. The patient had severe generalized hyperkeratotic lesions on the palms, soles, and perioral skin as well as hyperkeratosis of oral mucosa at multiple sites.

This case report documents the first reported case of Olmsted syndrome to be associated with severe periodontal disease. The altered differentiation of oral mucosa linked to Olmsted syndrome may contribute to the periodontal disease. Patients diagnosed with this syndrome should receive a comprehensive oral examination to determine whether periodontal destruction is a significant component of their disease or not.